WHY DO THEY KEEP COMING BACK? PERSISTENT FREQUENT ATTENDERS IN PRIMARY CARE COLOFON This study was part of the perfactio group. This research on frequent attenders was funded by grants from the Academic Medical Center, University of Amsterdam, the Netherlands; Stichting steunfonds medische en sociale dienstverlening, Reigersbos (Foundation to support medical and social services Reigersbos); and a grant from the Netherlands Organization for Health Research and Development (ZonMw), programma Alledaagse ziekten (common diseases; nr 42011002). Copyright 2014 Frans Smits. All rights reserved. No part of this thesis may be reproduced or transmitted, in any form or by any means, without the prior permission of the author. ISBN: Cover image: Graphic design: Printed by: 978-90-6464-808-3 Unknown photographer, waiting room Laura Smits GVO drukkers & vormgevers B.V, www.proefschriften.nl WHY DO THEY KEEP COMING BACK? PERSISTENT FREQUENT ATTENDERS IN PRIMARY CARE ACADEMISCH PROEFSCHRIFT ter verkrijging van de graad van doctor aan de Universiteit van Amsterdam op gezag van de Rector Magnificus prof. dr. D.C. van den Boom ten overstaan van een door het college voor promoties ingestelde commissie, in het openbaar te verdedigen in de Agnietenkapel op donderdag 18 september 2014, te 14.00 uur door Franciscus Thomas Maria Smits geboren te Breukelen PROMOTIE COMMISSIE Promotores: Prof. Dr. H. C. van Weert Prof. Dr. A.H. Schene Co-promotores: Dr. G. ter Riet Dr. J. Bosmans Overige leden: Prof. Dr. J.C.J.M. de Haes Prof. Dr. H.E. van der Horst Prof. Dr. A.L.M. Lagro-Janssen Prof. Dr M. Maas Dr. H.G. Ruhé Prof. dr. K. Stronks Faculteit der Geneeskunde “Wherever the art of Medicine is loved, there is also a love of Humanity” HIPPOCRATES contents Chapter 1: General introduction 008 PART I: MAPPING FREQUENT ATTENDERS IN PRIMARY CARE 020 Chapter 2: Defining frequent attendance in general practice 020 Chapter 3: Epidemiology of (persistent) frequent attenders A 3-y historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders- 030 Chapter 4: Is persistent frequent attendance predictable? A historic 3-year cohort study 044 Chapter 5: Predictability of persistent frequent attendance in primary care: A temporal and geographical validation study 058 Chapter 6: Morbidity and doctor characteristics only partly explain the substantial healthcare expenditures of frequent attenders - A record linkage study between patient data and reimbursements data - 076 PART II: REVIEW OF THE LITERATURE ABOUT INTERVENTIONS ON FREQUENT ATTENDERS IN PRIMARY CARE Chapter 7: Interventions on frequent attenders in primary care. A systematic literature review 6 092 092 PART III: A PROSPECTIVE STUDY OF FREQUENT ATTENDERS 106 Chapter 8: Why do they keep coming back? -Aetiology of persistence of frequent attendance in primary care; a prospective cohort study- 106 Chapter 9: Is treatment of psychiatric morbidity in frequent attenders cost-effective in comparison with usual general practitioner care? Results of a modelling study 130 Chapter 10: General discussion 152 Chapter 11: Appendices 164 Summary 178 Samenvatting 184 Dankwoord 190 Curriculum vitae 194 PhD portfolio 196 List of publications 198 7 general introduction Background and motivation for this study Every General Practitioner (GP) will recognize the slight confusion when the name of a patient that recently visited the practice several times, is again on the daily schedule: “A new appointment again? Didn’t we reach a conclusion last time? Did the complaints aggravate? Was the diagnosis and intervention (perceived as) insufficient?” Sometimes you feel you have fallen a bit short, annoyed or unable to think of a good explanation. And you may wonder whether there might be something in this patient that you did not recognize and, consequently, did not treat adequately, resulting in this repeated attendance. Patients who visit their GP much more often than other patients in the practice are called Frequent Attenders (FAs). In this thesis we describe (persistent) FAs and investigate factors leading to (persistence of) frequent attendance, relations with the wellbeing of these patients, treatment options for FAs and cost consequences. Most patients only attend their GP frequently for a short period of time1-4 and only 20–30% of FAs continues to attend frequently in the following year.1-3 FAs not only frequently attend their GP, but they are also more often referred to specialist care than non-frequent attenders (non-FAs).5 The burden on primary and secondary care of FAs is high. In the United Kingdom approximately 80% of a GP’s clinical work is spent on 20% of his/her patients, and this often leads to care that is not effective in helping the patient.6 Most short-term frequent attendance can adequately be explained, for example by a temporary medical problem. However, when frequent attendance spans more (consecutive) years both chronic physical and long-lasting psychosocial problems are often present in this group of patients.7-11 Psychological distress, low physical quality of life (QoL), and a low educational level are associated 8 WHY DO THEY KEEP COMING BACK? with persistent frequent attending.9;12 Persistent frequent attendance may be considered an easily detectable type of behavior, indicating underlying psychosocial or psychiatric problems and low QoL, sometimes undetected and untreated. Therefore, we concentrate this research on persistent frequent attendance. Because somatic problems are already adequately addressed in everyday GP-care and (chronic) care models, we more specifically focus on the role of psychological and social factors in the aetiology of (persistence of) frequent attendance. We think this approach might also contribute to the thoughtful design of preventive strategies for persistence of frequent attendance. Definition of (persistent) frequent attenders and how to select FAs in a normal GP’s practice? Several methods for selecting frequent attenders have been used until now.13;14 The older the patient, the more frequently he or she will visit healthcare professionals in primary and secondary care, just because of physical aging.15 Women of all age groups make more use of healthcare than men.11;16 Consultation frequencies differ between countries and, within countries, by GP.14 Just selecting the most attending persons thus will lead to the selection of predominantly older females and patients of a limited number of GP’s. Hence, it is more appropriate to select the exceptional users within every age and sex group of a GP (practice).14 Such a proportional threshold definition selects the exceptional users compared to their peers and allows for meaningful comparison between individual practices, periods, and countries. Therefore, in this thesis we define FAs as those patients whose attendance rate ranks in the top 10 centile per age and sex group in a GP-practice within a time frame of one year, and persistent FAs as those patients who are FA during three consecutive years. To calculate FAs we used all face-to-face consultations with GPs (consultations in the surgery and house-calls) and the number of all enlisted patients in a practice. Selecting FAs by using age groups with a small range (for example a 10-year range) is difficult and labour-intensive, especially in smaller populations like those of a single practice. It may result in low numbers of patients in each age group. In the UK, Howe advised using the following method to define FAs: group all men and for women create two age groups and calculate the top 10% attenders in each of these three groups.13 To establish which method would be most appropriate and feasible to use in the Netherlands, we tested different methods of selecting FAs in an average general (group) practice (Chapter 2). CHAPTER 1 9 How to understand and interpret frequent attendance? To structure our research we hypothesized that within the context of a given western GP-centred primary healthcare system, attendance rates may be influenced by patient characteristics (including morbidity), by GP characteristics (like work style, experience, personality and professional interests), and thirdly by the interpersonal dynamics between patients and their physicians (see figure 1). We restrict our scope to healthcare systems with a well-organized primary care in which GPs provide continuity of care for enlisted patients and act as gatekeepers to specialist care. Patient characteristics Most studies describing patient characteristics concern short-term frequent attenders. The decision whether to consult a GP seems to depend on the patient’s past experience with healthcare, the perception of the symptoms, the perception of the GP’s role as well as the relationship with the GP.17-20 Other reported factors are health anxiety (balancing fears), passivity, lack of control or mastery and mental health problems.20 One study found that FAs are often not aware of their frequent attendance. The interviewed patients regarded the GP as an appropriate figure to solve their distinctive and multiple physical symptoms and, despite their trust in the GP, some dissatisfaction with the (not) given treatment remained.21 FAs with medically unexplained symptoms (MUS) seem to persist in frequent attendance because of high health anxiety and concern about a missed diagnosis, often despite some level of insight in their condition.19 In the attachment theory cognitive schemas based on earlier repeated experiences with caregivers are considered to influence how individuals perceive and act within interpersonal relationships. The ‘insecure attachment style’ was shown to be associated with frequent attendance after adjusting for socio-demographic characteristics, presence of chronic physical illness and baseline physical function. The ‘preoccupied attachment style’ was associated with high primary care costs and utilization.22;23 These associations were particularly strong for those patients who believed that a physical problem caused their unexplained symptoms. High consultation rates may be conceptualized as pathological care-seeking behaviour linked to insecure attachment.23 Understanding frequent attendance as driven by difficulties in relating to care giving figures may help doctors to manage their frequently attending patients in a different way. Other authors observed that attendance rates depend on early child experiences and that families tend to be consistent in illness and consultation patterns over the years and even over the generations.17;18;24;25 10 WHY DO THEY KEEP COMING BACK? Figure 1. Theoretical model of possible aetiological explanations of persistence of Frequent attendance Life events Financial circumstances; educational level Somatic problems Demographic issues (work, living circumstances) Psychiatric Problems (axis I) Consultation frequency of the patient Family; Ethnicity Personality (axis II) Coping style (mastery) Communication between the patient and the GP; Somatic fixation? General practitioner Age /Sex Number of enlisted patients Habits in registering problems in the EMD Experience as a GP Corrected number of consultations per patient Special interests CHAPTER 1 11 GP determinants It seems plausible that GPs also play a role in the attendance frequency of their patients. Physicians differ significantly in their clinical decision-making. The mean consultation frequency, but also the number of lab tests and referrals to secondary care vary considerably between GPs and practices.26;27 However, little is known about the impact of GP specific determinants on the frequency of consultation and on persistent frequent attendance in some patients.28 A qualitative study described the emotions and thoughts of physicians at primary healthcare centers in Spain during consultations with short-term frequent attenders.29 Positive emotions regarding FAs were associated with young age of the physician and presence of the thought “This patient really needs me”. Feelings of lack of control were associated with working in rural centres and with negative thoughts about FAs. Anxious thoughts of the GP were associated with greater workload, more requests for tests, more requests to see the doctor outside regular hours, and negative thoughts about FAs. Guilt feelings were associated with a lower perceived ability to solve the patient’s problem, and with a poor physician-patient relationship. Sadness of the GP was associated with more frequent referrals to specialists.29 Interaction between patient and GP In the 1980s, some authors postulated that inadequate interpersonal dynamics between patients and their GP could cause more inappropriate and unnecessary consultations, testing and treatments, a phenomenon then labelled as “somatic fixation”.30-32 They emphasized the importance of adequate communication skills of the GP to break the chain of this fixation. However, literature describing the interaction between FAs and the GP is scarce.29 (Medical) problems of frequent attenders Most literature on FAs originates from countries which organise primary care through a system in which a primary care physician (e.g. a GP) serves a fixed group of enlisted patients. Apparently, frequent attendance is considered more of a problem in this healthcare system because payment of the GP is (largely) per enlisted patient and less per consultation. Several reviews from Scandinavian countries14, the United Kingdom33, Spain34 and Health Maintenance Organizations(HMO) in the United States35;36 describe morbidity of FAs during a one year period (1yFAs). In 1yFAs combinations of somatic and psychosocial problems are often observed and high rates of both psychological distress and psychiatric disorders are found.37-39 Rates of somatization among FAs vary between 16 and 45% .35;40;41 As far as we know there is only one study describing frequent 12 WHY DO THEY KEEP COMING BACK? attendance in the Netherlands42 and only one study from a country with an open access system for primary care (France). The latter found that, when adjusting for confounders, among four psychiatric diagnoses investigated, only somatoform disorders remained significantly associated with frequent attendance. Physical health and chronic diseases were not associated with frequent attendance.43 Thus, 1yFAs suffer more often from chronic somatic diseases, medically unexplained symptoms, psychiatric problems (e.g. depression, anxiety) and social problems than non-FAs.8;14 Less is known about persistent FAs, but existing evidence indicates that these patients not only suffer from more somatic, but, in particular, from more psychiatric problems.44;45 FAs who are depressed are more likely to continue to be high-utilizers than non-depressed FAs.46 Therefore, we examined the somatic and psychosocial morbidity of (persistent) frequent attenders (chapter 3). Workload and costs of FAs in primary and specialist healthcare FAs are more frequently referred by their GP to specialist care than non-frequent attenders (non-FAs).5 However, little is known about the magnitude of the differences in primary and specialist healthcare utilisation and costs between nonFAs and FAs, as well as between subgroups of FAs (short-term versus persistent FAs). Differences in workload and healthcare costs may be explained by the specific characteristics and morbidities of FAs, and by physician characteristics. If not, detection and treatment of underlying, not yet detected, conditions in FAs may result in less morbidity, a better quality of life and decrease in costs. Therefore, we examined the workload caused by FAs in primary care and costs of healthcare of (persistent) FAs in primary and specialist care and whether these costs can be explained by patients’ morbidities and by GP characteristics (chapter 3 and 6). Prediction of persistent frequent attendance Development of effective interventions to prevent 1yFAs to become persistent or repetitive FAs is only possible if knowledge is available about determinants that predict which one-year FAs are likely to become persistent FAs. However, literature about determinants that predict persistent frequent attendance is inconsistent and its interpretation is hampered by methodological differences (e.g. aetiological and causal versus predictive non-causal outlooks, or confusion of these two), and by different definitions of frequent attendance (e.g. proportional versus fixed cutoff). In prospective cohort studies, using a proportional definition (the upper 10%), low physical quality of life, low educational level12 and psychological distress (Hopkins Symptom Check List and Whiteley-7)9 predicted persistence of frequent CHAPTER 1 13 attendance over the next two consecutive years. Using a fixed cutoff definition of FA, one prospective cohort study found that female gender, obesity, former frequent attendance, fear of death, alcohol abstinence, low satisfaction, and irritable bowel syndrome were risk factors for persistence of frequent attendance during at least 3 out of the four next years.47 Another prospective cohort study concluded that the Ambulatory Diagnosis Groups “unstable chronic medical conditions”, “see and reassure conditions”, “minor time-limited psychosocial conditions”, and “minor signs and symptoms” predicted persistence of frequent primary care use the next year.48 A prediction rule may help GPs to identify which 1yFA is at risk to become a persistent FA using information from the electronic medical record. Such a rule, in addition to being clinically important, may also support the selection of more homogeneous patient groups in future randomized trials among (subgroups of) persistent frequent attenders (chapter 4 and 5). Attempts to support and help (persistent) FAs, and to lower attendance rates Is it possible to reduce the morbidity and use of healthcare of FAs and to improve their quality of life? Several RCTs evaluating interventions for FAs have been published but a clear overview of the different kind of interventions, an assessment of their quality and the results of the interventions is lacking.44;49-54 Therefore, we reviewed the literature to determine possible positive interventions to improve the morbidity and the quality of life and to lower attendance rate of FAs (chapter 7). This PhD thesis aims to answer these questions based on studies conducted among (persistent) frequent attenders in the Netherlands. Hereafter, the Persistent Frequent Attenders Risk Factors and treatment options (PERFACTIO) study is described and the structure of the thesis is outlined. The PERFACTIO study Part I: Mapping frequent attenders in primary care The first objective of this thesis was to establish the best method for selecting FAs in a normal practice setting in the Netherlands. Secondly, we wanted to study morbidity and GP’s workload of FAs of different duration. Thirdly, we examined whether it would be feasible, using information readily available in GPs’ electronic medical records, to predict which 1yFAs continue to attend frequently and whether this prediction (rule) could be validated in another setting and timeframe. Finally, we examined the costs of healthcare utilization by FAs of different duration in primary and specialist care and to explore whether these costs could be explained by the excess morbidity these FAs have or the characteristics of the GP’s. 14 WHY DO THEY KEEP COMING BACK? Research questions 1. What is the most feasible method to select FAs in a normal GP practice setting using the proportional definition? To answer this question we analysed in chapter 2 the data of the second Dutch National survey of General Practice. These data were collected over a oneyear period on health and healthcare-related behaviour from 375 899 persons, registered within 104 practices. We compared the quality of different FA selection methods in general practice in the Netherlands. 2. Which somatic, psychological and social problems do (persistent) FAs have? What are the differences between short-term and persistent FAs in this respect? What is the workload of a GP caused by (persistent) FAs? In chapter 3 we analysed the GP database of the Academic Medical Center, University of Amsterdam (Hag-net-AMC) of three consecutive years crosssectionally. We compared the diagnoses as registered by the GPs on the so-called Problem Lists of frequent attenders during none, one, two and three years, respectively. 3. Which readily available information noted by the GP in patients’ Electronic Medical Record predicts persistence of frequent attendance? In order to answer this question we performed in chapter 4 a historic threeyear cohort study (2003-2005). We analysed which readily available data of 1yFAs out of the Electronic Medical Record of the GPs predict persistence of frequent attendance. 4. Can the prediction rule developed in chapter 4 be validated in another time frame in the same GP database and in another GP database? In chapter 5 we performed a geographical and temporal validation of our prior prediction rule with data of a GP network in Eindhoven, the Netherlands (SMILE; geographical validation) and our own network (Hag-net-AMC) in another time frame (2009-2011). 5. Are FAs of primary care also high users of specialist care? What are the costs of healthcare of FAs in primary and specialist care? 6. Are the high costs in primary and specialist healthcare of FAs of different duration associated with patient’s morbidities and GP characteristics? In order to answer research question 5 and 6 we linked in chapter 6 clinical data of primary care patients to financial reimbursement data of the main health insurer of our region (healthcare expenditures in primary and specialist care), and GP characteristics. In a multilevel regression model, we analysed CHAPTER 1 15 the healthcare expenditures of FAs and whether these expenditures can be explained by the morbidity of the patients and the characteristics of their GPs. Part II: Review of the literature about interventions on frequent attenders in primary care. 7. Research question 7. To determine possible effective interventions to improve quality of life and lower attendance rate of FAs we systematically reviewed the literature about possible effective interventions in FAs in chapter 7. Part III: A prospective study of frequent attenders The objective of the prospective study was to better understand the causes of persistence of frequent attendance. This may facilitate the rational selection of diagnostic tests and (better) prevention strategies. Potentially effective interventions should be based on these aetiological factors for persistence of frequent attendance. Research question eight is: 8. Which (in particular psychosocial) factors are associated with persistence of frequent attendance in a prospective cohort of 1-year FAs? Is there a supraadditive effect of combinations of somatic, psychological and socials factors? In order to answer these questions we conducted a prospective cohort study of 1yFAs and collected data about their GPs in chapter 8. With a multilevel regression analysis we evaluated which patient and GP characteristics are associated with persistence of frequent attendance. Screening and consecutive treatment of patients in primary care tends to have disappointing results.55-58 Therefore, we extrapolated the findings from the cohort study among FAs over a period of 5 years and combined this with potential treatment effects using modelling techniques. Research question nine is: 9. To evaluate whether systematic detection and treatment of depression and anxiety after one or two years of frequent attendance may be cost-effective compared to usual GP care. Therefore we performed in chapter 9 a Cost Effectiveness Analysis (CEA) with data of the cohort of chapter eight. With a Markov simulation we analysed whether diagnosing and treating of depression and anxiety (as measured by the Patient Health Questionnaire) in FAs might be cost effective after one or two years of frequent attendance. We end the thesis with chapter 10, which provides a general discussion and conclusion of our research. 16 WHY DO THEY KEEP COMING BACK? 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An assessment of the attributes of frequent attenders to general practice. Family Practice 1998; 15(3):198-204. (39) Jyvasjarvi S, Joukamaa M, Vaisanen E, Larivaara P, Kivela S, KeinanenKiukaanniemi S. Somatizing frequent attenders in primary health care. J Psychosom Res 2001; 50(4):185-192. (40) Karlsson H, Joukamaa M, Lahti I, Lehtinen V, Kokki-Saarinen T. Frequent attender profiles: different clinical subgroups among frequent attender patients in primary care. J Psychosom Res 1997; 42(2):157-166. WHY DO THEY KEEP COMING BACK? (41) De Waal MWM, Arnold IA, Eekhof JAH, van Hemert AM. Somatoform disorders in general practice. Br J Psych 2004; 184:470-476. (42) van der Ploeg HM. Persoonlijkheid en medische consumptie: Een onderzoek naar de relatie van persoonlijkheidsfactoren en de frequentie van huisartsbezoek. Swets &Zeitlinger B.V.; 1980. (43) Norton J, David M, de Roquefeuil G, Boulenger JP, Car J, Ritchie K et al. Frequent attendance in family practice and common mental disorders in an open access health care system. J Psychosom Res 2012; 2012(6):413-418. (44) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (45) Pearson SD, Katzelnick DJ, Simon GE, Manning WG, Helstad CP, Henk HJ. Depression among high utilizers of medical care. J Gen Intern Med 1999; 14(8):461-468. (46) Henk HJ, Katzelnick DJ, Kobak KA, Greist JH, Jefferson JW. Medical costs attributed to depression among patients with a history of high medical expenses in a health maintenance organization. Arch Gen Psychiatry 1996; 1996(10):899904. (47) Koskela TH, Ryynanen OP, Soini EJ. Risk factors for persistent frequent use of the primary health care services among frequent attenders: a Bayesian approach. Scand J Prim Health Care 2010; 28(1):55-61. (48) Naessens JM, Baird MA, Van Houten HK, Vanness DJ, Campbell CR. Predicting persistently high primary care use. Ann Fam Med 2005(4):324-330. (49) Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ, Helstad CS. Cost-effectiveness of systematic depression treatment for high utilizers of general medical care. Arch Gen Psychiatry 2001; 58(2):181-187. (51) Christensen MB, Christensen B, Mortensen JT, Olesen F. Intervention among frequent attenders of the outof-hours service: A stratified cluster randomized controlled trial. Scandinavian Journal of Primary Health Care 2004; 22(3):180-186. (52) Katon W, Von KM, Lin E, Bush T, Russo J, Lipscomb P et al. A randomized trial of psychiatric consultation with distressed high utilizers. Gen Hosp Psychiatry 1992; 14(2):86-98. (53) Bellon JA, Rodriguez-Bayon A, de Dios LJ, Torres-Gonzalez F. Successful GP intervention with frequent attenders in primary care: randomised controlled trial. Br J Gen Pract 2008; 58(550):324330. (54) Barsky AJ, Ahern DK, Bauer MR, Nolido N, Orav EJ. A Randomized Trial of Treatments for High-Utilizing Somatizing Patients. J Gen Intern Med 2013; 2013. (55) Baas KD, Wittkampf KA, van Weert HC, Lucassen P, Huyser J, van den Hoogen H et al. Screening for depression in high-risk groups: prospective cohort study in general practice. The British Journal of Psychiatry 2009; 194(5):399403. (56) Bosmans J, Schreuders B, van Marwijk H, Smit J, van Oppen P, van Tulder M. Cost-effectiveness of problem-solving treatment in comparison with usual care for primary care patients with mental health problems: a randomized trial. BMC Family Practice 2012; 13(1):98. (57) Bosmans JE, van Schaik DJ, de Bruijne MC, van Hout HP, van Marwijk HW, van Tulder MW et al. Are psychological treatments for depression in primary care cost-effective? J Ment Health Policy Econ 2008; 11(1):3-15. (58) Thombs BD, Ziegelstein RC, Roseman M, Kloda LA, Ioannidis JP. There are no randomized controlled trials that support the United States Preventive Services Task Force guideline on screening for depression in primary care: a systematic review. BMC Med 2014:13-12. (50) Adam P, Brandenburg DL, Bremer KL, Nordstrom DL. Effects of team care of frequent attenders on patients and physicians. Fam Syst Health. 2010 Sep;28(3):247-57. GENERAL INTRODUCTION 19 part I MAPPING FREQUENT ATTENDERS IN PRIMARY CARE chapter 2 DEFINING FREQUENT ATTENDANCE IN GENERAL PRACTICE Frans Th. M. Smits, Jacob Mohrs, Ellen E. Beem, Patrick J.E. Bindels, Henk C.P.M. van Weert BMC Family practice, 2008, 9:21. ABSTRACT Background General practitioners (GPs) or researchers sometimes need to identify frequent attenders (FAs) in order to screen them for unidentified problems and to test specific interventions. We wanted to assess different methods for selecting FAs to identify the most feasible and effective one for use in a general (group) practice. Methods In the second Dutch National Survey of General Practice, data were collected on 375 899 persons registered with 104 practices. Frequent attendance is defined as the top 3% and 10% of enlisted patients in each one-year age-sex group measured during the study year. We used these two selections as our reference standard. We also selected the top 3% and 10% FAs (90 and 97 percentile) based on four selection methods of diminishing preciseness. We compared the test characteristics of these four methods. Results Of all enlisted patients, 24 % did not consult the practice during the study year. The mean number of contacts in the top 10% FAs increased in men from 5.8 (age 15-24 years) to 17.5 (age 6475 years) and in women from 9.7 to 19.8. In the top 3% of FAs, contacts increased in men from 9.2 to 24.5 and in women from 14 to 27.8. The selection of FAs becomes more precise when smaller age classes are used. All selection methods show acceptable results (kappa 0.849 – 0.942) except the three group method. Conclusion To correctly identify frequent attenders in general practice, we recommend dividing patients into at least three age groups per sex. 22 WHY DO THEY KEEP COMING BACK? Background In primary care, the workload of General Practitioners (GPs) is significantly related to a minority of patients who consult more frequently than their peers1. Studies are consistent in confirming that these frequent attenders (FAs) have high rates of physical disease, psychiatric illness, social difficulties and emotional distress2-4. Because frequent attendance can be related to undisclosed medical problems, identifying FAs could help GPs to select those patients who may need an adjustment to the care they receive5. The combination of large workload and high rate of (chronic) disease make FAs an important group for a GP not only to study but also to treat. Exceptional attendance is also considered as an indicator of inappropriate consulting behaviour and healthcare use6-11. Health services research has therefore used frequent attendance for identifying both inadequate health care delivery and possible misuse of health care. Trials on the effect of (mainly psychiatric) interventions on the attendance rate and morbidity of FAs showed conflicting results12-15. In a review of interventions on FAs we found indications that frequent attendance might be a sign of as yet undiagnosed major depressive disorder (MDD) and that treatment of MDD might improve the depressive symptoms and the quality of life of depressed FAs. We found no evidence that it is possible to influence healthcare utilization12-15. The interpretation of studies on frequent attendance is hampered because of differences in the organisation of health care, the setting and the definition of FAs. Age and sex have been shown to be highly associated with the frequency of attendance16. Selecting FAs without adjusting for age and sex will predominantly result in the selection of older women3. Therefore, any study of frequent attendance requires a clear definition of these patients and a clear description of the selection process. After reviewing the literature on frequent attendance, Vedsted suggested that frequent attendance should be defined as a proportional part (highest 10%) of all attenders, stratified for age and sex17. Selecting FAs by using age groups with a small band (for instance ten-year groupings) is difficult, especially in smaller populations like those of a (group) practice because of the resulting low number of patients in each cell. Therefore, Howe et al. developed an easy cohort definition to identify those patients whose attendance patterns are unusual for their sex and age. She stated that, dividing the male population into two different age groups (15-44; 45-74 years), would result in including 95% of the total male patients identified as attending at or above the 97th percentile compared with the more complex procedure of ten-year groupings. Further, she concluded that no such division of the population was needed for females, as their consultation rates were considered fairly constant. She advised further analysis on the validity of this method in other populations to be performed18. In the Netherlands every citizen is enlisted by one GP and Dutch inhabitants consult their own GP for all medical complaints. CHAPTER 2 23 The GP functions as a gatekeeper for specialist care. GP-care in 2001 was paid either by a social sick-fund or an obligatory private insurance. We used the large database of the second Dutch National Survey of General Practice (2001) as a unique possibility for comparing the quality of different FA selection methods in general practice in the Netherlands. Our aim was to assess these methods and to identify the most feasible and effective one for use in an average general (group) practice. Method In the second Dutch National survey of General Practice, data were collected over a one-year period on health and healthcare-related behaviour from 375 899 persons, registered with 104 practices. Eight practices were excluded because of insufficient data (see flow diagram). Population, practices and GPs were representative for the Dutch population, with a slight under-representation of single-handed GPs. The study design, methods, response and quality of the data of this extensive second Dutch National Survey have been published elsewhere in more detail19-21. To correct for loss or growth of the practices involved during the study year, we used the data of patients enlisted within each practice over the complete one-year period (n= 263 148) as the denominator. As most previous studies on frequent attendance have excluded children and the very old, we also only used the data of patients between the ages of 15 and 74 years. Figure 1. Flow diagram National Survey of General Practice (NS2). • 375 899 patients • 104 general practices NS2 • N= 276 924 patients • 96 general practices Exclusion: < 15 y; >75 y. 8 practices because of insufficient data. Exclusion: Not enlisted all year. Minus 13 776 Study population: • N= 263 148 patients Analyses 24 WHY DO THEY KEEP COMING BACK? For all patients included, each contact with the primary care team (consultations, house calls and telephone calls) was registered. We calculated the contact frequencies of all patients between the ages of 15-74 years for every combination of age and sex. As in previous research, the top 3% and the top 10% consulting patients from this calculation were defined as FAs. We also included patients with no attendance. These two selections were then used as our reference standard. As index-selections, we selected the top 3% and 10% of FAs of the same population by dividing the genders into four different age group clusters ranging from just one to as many as six. We compared the sensitivity and specificity of the selection criteria in each of these four cluster groups: 1. Per each 10-year age band: 6 classes per sex category17;18. 2. Per each 15-year age band: 4 classes per sex category. 3. According to the sex-age grouping, used by the WONCA classification committee, we tested an adjusted selection method with 3 age classes per sex category: 15-44 years, 45-64 years and 65-74 years22. 4. Dividing males into two separate cohorts (15-44 years; 45-74 years) and all women in one cohort: the three group method18. By constructing four by four tables we calculated the test characteristics (the sensitivity, specificity, positive and negative predictive value and kappa) of these four clusters, using the one-year age band method as reference standard. All data were analysed using SPSS 14.0 for windows. The study was conducted according to the Dutch legislation on data protection (Ministry of Justice, the Netherlands) Results From the total number of enlisted patients, 63102 (24%) of which 21 090 female and 42 012 men did not consult their primary care practice during the study year. Women consulted more frequently than men and older age correlated with a rising number of contacts for both sexes (figure 1). The mean number of contacts increased in men from 1.62 (age 15-24 years) to 5.13 (age 65-74 years) and in women from 3.32 (age 14-24 years) to 6.27 (65-74 years). The mean attendance by sex and age of the top 3% and 10% attenders is presented in figure 1. The mean number of contacts in the top 10% FAs increased in men from 5.84 (age 15-24 years) to 17.46 (age 64-75 years) and in women from 9.72 (age 15-24 years) to 19.83 (age 64-75 years). The mean number of contacts in the top 3% of FAs increased in men from 9.21 (age 15-24 years) to 24.52 (age 64-75 years) and in women from 14.02 (age 15-24 years) to 27.83 (age 64-75 years). All test characteristics (sensitivity, specificity, positive and negative predictive value and kappa), are summarized in table 1. With 6 classes, the kappa is 0.942 (10 % FA) and 0.925 (3% FA) but with the 3 group-method the kappa is 0.818 (10% FA) and 0.756 (3% FA). Test characteristics improve with smaller age classes and CHAPTER 2 25 logically sensitivity drops by using the three group method, even more in females than in males. The test characteristics are slightly better when the top 10 % is selected instead of the top 3% of FAs. All methods show acceptable results (kappa 0.849 - 0.942) except the three group method. Discussion The purpose of this study was to compare different methods for selecting frequent attenders in primary care and to identify the most feasible method with acceptable test characteristics in a general (group) practice . We found specificity to be about the same in all methods, but sensitivity diminishes gradually when larger age groupings are used and shows a drop in the three group method. This means that with the three group method (3% resp.10% FA) 25% resp. 17 % of the FAs will not be identified. For instance selecting the top 10% of FAs the three group method misses 5247 FAs (17%) of which 58% female and 47% in the age between 15 and 24. This study is the first attempt to compare different methods of identifying FAs. In a large database like the Dutch National Survey, the reference method (with one year sex-age bands) is the most precise method for identifying FAs. In smaller databases however, such a method results in very few patients within each age band and is therefore not feasible. Our results demonstrate that specificity and sensitivity for identifying FAs increases when smaller age groups are used, as could be expected. On the level of a general (group) practice, less precise methods can be used with Figure 2. Mean attendance per sex, all attenders and the top 3%/10% attenders 30 mean attendance 3% FA women mean attendance 3% FA men 25 mean attendance 10% FA women 20 mean attendance 10% FA men number of contacts 15 10 mean attendance women mean attendance men 5 0 15-24 25-34 35-44 45-54 10-year age group 26 WHY DO THEY KEEP COMING BACK? 55-64 65-74 Table 1. Overview of the test characteristics of the four selection methods, 6 cl, method 3%FA1 6 cl, method 10%FA 2 4 cl, method 3%FA3 4 cl, method 10%FA4 women men women men women men women men Sensitivity 94,2 89,4 94,0 93,1 89,4 93,6 94,7 91,5 Specificity 99,8 99,8 99,3 99,8 99,8 99,6 99,6 99,1 5 93,7 93,7 94,3 98,2 93,7 89,5 96,3 93,1 NPV6 99,8 99,6 99,2 99,0 99,6 99,8 99,3 98,8 Kappa 0,937 0,912 0,934 0,950 0,912 0,911 0,949 0,912 PPV 3 cl, method 3%FA7 6 cl, method 10%FA8 4 cl, method 3%FA9 4 cl, method 10%FA10 women men women men women men women men Sensitivity 89,0 84,8 96,0 85,5 76,1 73,6 79,3 86,5 Specificity 99,3 99,3 98,3 98,8 99,1 99,4 98,5 97,6 PPV 88,1 80,4 87,8 90,6 74,1 82,3 86,9 83,1 NPV 99,6 99,5 99,5 98,0 99,2 99,1 97,4 98,1 Kappa 0,881 0,819 0,906 0,864 0,742 0,770 0,809 0,826 Table 2. Men and women 6 cl, method 4 cl, method 3 cl, method 3 group-method 3% FA 10% FA 3% FA 10% FA 3% FA 10% FA 3% FA 10% FA Sensitivity 91,8 93,5 90,9 93,0 86,9 90,6 74,8 83,0 Specificity 99,8 99,5 99,7 99,3 99,4 98,5 99,3 98,0 PPV 93,7 96,3 91,4 94,6 84,1 89,1 78,0 84,8 NPV 99,7 99,1 99,7 99,1 99,5 98,7 99,1 97,7 kappa 0,925 0,942 0,912 0,930 0,849 0,885 0,756 0,818 1. 2. 3. 4. 5. 6. 7. 8. 9. 6 cl. method: selection FAs per 10 years of age. Idem 4 cl. method: selection of FAs per 15 years of age Idem positive predictive value negative predictive value 3 cl. method: selection of FAs in 3 age groups (15-44; 45-64; 65-74). Idem 3 group method: selection of male FAs in two age groups (15-44; 45-74) and women in one group. 10. Idem CHAPTER 2 27 acceptable results: for instance, by dividing all patients into at least 3 age cohorts per sex. For studies on larger patient groups, it is best to use the smallest possible age groupings, mainly for reasons of positive predictive value. Standardisation of methods for selecting FAs is needed in order to allow comparisons between studies to take place. The purpose for which FAs need to be selected, as well as the limitations of the database, can determine the degree of the desired precision. For example, if a GP wants to use frequent attending as a red flag pointing at unidentified medical problems, it would not be too big a problem to incorrectly select a patient (false positive). Not selecting an FA (false negative) seems to be a bigger problem, but the negative predictive value is high in all methods. However researchers have to use the smallest age band possible to correctly select FAs. Conclusion We conclude that in order to identify exceptional users of health care, sex and age have to be taken into account. The best method for identifying frequent attenders is to use small age and sex groups. If this is not possible or needed, for instance in a single general (group) practice, we recommend that GPs divide their patients into at least 3 age groups per sex category in order to identify their exceptional attenders. 28 References (1) Neal RD, Heywood PL, Morley S, Clayden AD, Dowell AC. Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br J Gen Pract 1998; 48(426):895-898. (2) Gill D, Sharpe M. Frequent consulters in general practice: a systematic review of studies of prevalence, associations and outcome. J Psychosom Res 1999; 47(2):115-130. (3) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (4) de Waal MW, Arnold IA, Eekhof JA, Assendelft WJ, van Hemert AM. Followup study on health care use of patients with somatoform, anxiety and depressive disorders in primary care. BMC Fam Pract 2008; 9(1):5. (5) Karlsson H, Joukamaa M, Lahti I, Lehtinen V, Kokki-Saarinen T. Frequent attender profiles: different clinical subgroups among frequent attender patients in primary care. J Psychosom Res 1997; 42(2):157-166. (6) Booth BM, Ludke RL, Wakefield DS, Kern DC, du Mond CE. Relationship between inappropriate admissions and days of care: implications for utilization management. Hosp Health Serv Adm 1991; 36(3):421-437. (7) Brandon WR, Chambers R. Reducing emergency department visits among high-using patients. J Fam Pract 2003; 52(8):637-640. (8) Hansagi H, Olsson M, Sjoberg S, Tomson Y, Goransson S. Frequent use of the hospital emergency department is indicative of high use of other health care services. Ann Emerg Med 2001; 37(6):561-567. (9) Kapur K, Young AS, Murata D, Sullivan G, Koegel P. The economic impact of capitated care for high utilizers of public mental health services: the Los Angeles PARTNERS program experience. J Behav Health Serv Res 1999; 26(4):416429. WHY DO THEY KEEP COMING BACK? (10) Reid S, Wessely S, Crayford T, Hotopf M. Frequent attenders with medically unexplained symptoms: Service use and costs in secondary care. British Journal of Psychiatry 2002; 180(3):248-253. (11) Stewart P, O’Dowd T. Clinically inexplicable frequent attenders in general practice. Br J Gen Pract 2002; 52(485):1000-1001. (12) Christensen MB, Christensen B, Mortensen JT, Olesen F. Intervention among frequent attenders of the outof-hours service: a stratified cluster randomized controlled trial. Scand J Prim Health Care 2004; 22(3):180-186. (13) Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ, Helstad CP. Costeffectiveness of systematic depression treatment for high utilizers of general medical care. Archives of General Psychiatry /2; 58(2):181-187. (19) Jones R, Schellevis F, Westert G. The changing face of primary care: the second Dutch national survey. Fam Pract 2004; 21(6):597-598. (20) Schellevis FG, Westert GP, De Bakker DH. [The actual role of general practice in the dutch health-care system. Results of the second dutch national survey of general practice]. Med Klin (Munich) 2005; 100(10):656-661. (21) Westert GP, Schellevis FG, De Bakker DH, Groenewegen PP, Bensing JM, van der ZJ. Monitoring health inequalities through general practice: the Second Dutch National Survey of General Practice. Eur J Public Health 2005; 15(1):59-65. (22) Lamberts H, Wood M e. International classification of primary care. Oxford: Oxford University Press; 1988. (14) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (15) Katon W, von Korff M, Lin E, Bush T. A randomized trial of psychiatric consultation with distressed high utilizers. General Hospital Psychiatry /3; 14(2):86-98Record. (16) Little P, Somerville J, Williamson I, Warner G, Moore M, Wiles R et al. Psychosocial, lifestyle, and health status variables in predicting high attendance among adults. Br J Gen Pract 2001; 51(473):987-994. (17) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (18) Howe A, Parry G, Pickvance D, Hockley B. Defining frequent attendance: evidence for routine age and sex correction in studies from primary care settings. Br J Gen Pract 2002; 52(480):561-562. CHAPTER 2 29 chapter 3 EPIDEMIOLOGY OF (PERSISTENT) FREQUENT ATTENDERS -A 3-YEAR HISTORIC COHORT STUDY COMPARING ATTENDANCE, MORBIDITY AND PRESCRIPTIONS OF ONE-YEAR AND PERSISTENT FREQUENT ATTENDERSFrans T. Smits, Henk J. Brouwer, Gerben ter Riet, Henk C. van Weert BMC Public Health 2009, 9:36 ABSTRACT Background Attendance rates for patients visiting a General Practitioner (GP) vary. Patients who remain in the top 10 centile of the age and sex adjusted attendance rate for at least 3 years are known as persistent frequent attenders. GPs spend a disproportionate amount of time on persistent Frequent Attenders. So far, trials on the effect of (mostly psychiatric) interventions on frequent attenders have shown negative results. However, these trials were conducted in short-term (< 3year) Frequent Attenders. It would be more reasonable and efficient to target diagnostic assessment and intervention at persistent Frequent Attenders. Typical characteristics of persistent Frequent Attenders, as distinct from frequent attenders during one year and nonFrequent Attenders, may generate hypotheses with respect to modifiable factors on which new randomized trials may be designed. Methods We used the data of all 28,860 adult patients from 5 primary healthcare centres, participating in a GP-based continuous morbidity registration network. Frequent Attenders were patients whose attendance rate ranked in the (age and sex adjusted) top 10 percent during 1 year (1- year-Frequent Attenders) or 3 years (persistent Frequent Attenders). All other patients on the register over the 3 year period were referred to as non-Frequent Attenders. The lists of current medical problems as registered and coded by the GP using the International Classification of Primary Care (ICPC) were used to assess morbidity. First, we determined which proportion of 1-year-Frequent Attenders was still a frequent attender during the next two consecutive years and calculated the GPs’ workload for these patients. Second, we compared the morbidity and the number of prescriptions for non-Frequent Attenders, 1-year-Frequent Attenders and persistent Frequent Attenders known to the GP. 32 WHY DO THEY KEEP COMING BACK? Results Of all 1yFAs, 15.4 % became a pFA (1.6% of all patients). Of the FAs, 3,045 (10.6%) were responsible for 39% of the faceto-face consultations; 470 patients who would become pFAs (1.6%) were responsible for 8% of all consultations in 2003. Compared to non-FAs and 1yFAs, considerable more social problems, feelings of anxiety, addictive behaviour and medically unexplained physical symptoms were seen in pFAs. FAs differ less where the prevalence of chronic somatic diseases - respiratory problems, cardiovascular problems and diabetes mellitus- are concerned. Conclusion One out of every seven 1-year-frequent attenders (15.4%) becomes a persistent Frequent Attender. Compared with nonFrequent Attenders, and 1-year-Frequent Attenders, persistent Frequent Attends consume more health care and are diagnosed not only with more somatic diseases but especially more social problems, psychiatric problems and medically unexplained physical symptoms. CHAPTER 3 33 Background General practitioners (GP) spend a large part of their time on a small proportion of their patients. It is estimated that about 80% of a GP’s clinical work is spent on 20% of their patients1. In most studies, frequent attendance is defined as an age and sexadjusted attendance rate ranking in the top 10 centile within a time frame of one year (1-year-Frequent Attenders)2;3. Systematic reviews show that these 1-year-Frequent Attenders are more likely to suffer from physical and psychiatric illness, social difficulties and emotional distress 2;4;5. High attendance rates are also found for patients with medically unexplained somatic symptoms, health anxiety and perceived poor health 5-7. In addition, frequent attendance may be a sign of inappropriate consultation behaviour 8-11. At this point, we should ask the question whether or not it is possible to treat frequent attenders and reduce their attendance rates? Trials on the effect of (mainly psychiatric) interventions have shown conflicting results 12;12. No study has shown convincing evidence that any intervention improves the quality of life or morbidity of Frequent Attenders in primary care, although there is some evidence that an effect might exist in a subgroup of Frequent Attenders – that of depressed patients. There is no evidence to suggest that the utilization of health care by Frequent Attenders can be influenced. The only trials that showed positive effects were with patients who were Frequent Attender over a period of two years; all others used a time frame of one year 13;14. 34 This means that these studies may have targeted the wrong group of transient Frequent Attenders. Until now most research on frequent attendance has been cross-sectional and used one-year attendance rates. The few longitudinal studies conducted showed regression of attendance to the mean in the longer run, with only 20-30% of FAs continuing to attend frequently in the following year 15-17. However, these studies on persistent frequent attendance used different definitions of FAs and lacked the power to detect differences in morbidity between transient and persistent frequent attenders. This study presents the results of a historic 3-year cohort study on 28,860 adult patients in a longitudinal primary care database. Our first objective was to study the natural course of frequent attendance and to determine the proportion of 1-year-Frequent Attenders who remain a Frequent Attender during two consecutive years and to calculate the GP workload for non-Frequent Attenders, 1-year-Frequent Attenders, and persistent Frequent Attenders. Secondly, we wanted to determine whether and how persistent Frequent Attenders differ from 1-yearFrequent Attenders and normal attenders. Methods Patient population Five primary healthcare centres in Amsterdam provided data for this study. These centres participate in the GP-based continuous morbidity registration network of the Department of General Practice WHY DO THEY KEEP COMING BACK? at the Academic Medical Centre of the University of Amsterdam. The studied patients have a lower socio-economic level and are of more non-western descent than the average Dutch population. The age distribution refers about the Dutch population. In this network, electronic medical record data are extracted for research purposes. The participating GPs use a problem oriented registration method. For this study we used the following data: the numbers of face-toface GP consultations, the lists of patients’ current medical problems as registered and coded by the GPs using the ICPC, the number of a selection of prescriptions for all enlisted patients from 1 January 2003 through 31 December 2005. Selection of 1yFAs, pFAs and non-FAs Frequent Attenders were defined as those patients whose attendance rate ranked nearest to the top 10th centile of their sex and age group (15-30 years; 31-45 years; 4660 years; 61 years+) 2 3. Frequent Attenders were determined for each of the years 2003, 2004 and 2005. As starting point, we took the one-year-frequent attenders for the year 2003. We defined persistent Frequent Attenders as those patients who continued to be a frequent attender over the three year period. Patients who were never a Frequent Attender in the three year study period (non-Frequent Attenders) were used as a reference group. We compared the three selections. Patients younger than 15 years were excluded, because their consultations often depend on their parents. A multivariable analysis was performed to check for selective loss to follow up. Attendance Only face-to-face consultations with GPs (consultations in the surgery and housecalls) were included. Consultations with other practice staff were excluded because these contacts are mostly initiated by the GP and relate mostly to the monitoring of chronic diseases. We determined the mean number of consultations per age and sex group for the three groups of patients. Morbidity In the problem oriented approach to medical record keeping, patients can have a list of current medical problems (“Problem list”). Different from the definition used in the UK, in the Netherlands a current medical problem is defined by the GP as: any medical problem (disease or complaint) which needs continual medical attention or monitoring; any complaint or disease presented to the GP that has lasted more than 6 months and/or any recurrent medical problem (more than 4 complaints per half year).Every problem on this list was coded by the GPs using the ICPC 18. Please see appendix 1 for a list of the ICPCcodes. The data from these problem lists were extracted at the end of 2003 and the end of 2005. The numerator in the prevalence calculations was the number of enlisted patients with a certain current problem at the end of the two periods. Thus the prevalence of each medical problem was calculated for 1-year Frequent Attenders at the end of the first year, for persistent and non-Frequent Attenders at the end of the third year. CHAPTER 3 35 Box 1. Approach to the multivariable analysis Loss to follow-up 368 patients (12%) were lost at some point over the two years of follow-up. We argued that, in theory, a potential frequent attender might move out of the practice due to dissatisfaction with care. The resulting selection bias may attenuate associations found between the selected indicators and frequent attendance. We tested our hypothesis in a multivariable logistic regression analysis with an indicator variable “1 = moved house” and “0 otherwise” as the dependent variable and 9 independent indicators (see below). Our hypothesis was not confirmed. On the contrary, we found some evidence that those with at least one chronic somatic illness were less likely to have moved out of the practice(odds ratio 0.73 (95%CI from 0.54 to 0.99)); all other associations were neither strong nor significant. These results support the view that important selection bias is unlikely. Sixty-eight patients (2.2%) had died over the two year follow-up period, but since, by definition, these patients cannot become 3-year frequent attenders, selection bias by death is impossible. Variable selection The 1. 2. 3. 4. 5. 6. 7. 8. 9. 9 candidate predictors, modelled as 11 variables, included: age at baseline (continuous), sex, number of problems on the problem list (continuous), any of the three chronic somatic illnesses just mentioned (yes/no), any psychological/social problem (yes/no), any medically unexplained physical problem (yes/no), psychoactive medication (yes/no), average monthly number of prescriptions for antibiotics (0 = reference category; 1-2; >2), average monthly number of prescriptions for analgesics (0 = reference category; 1-4; >4). Figure 1. Flow diagram: Persistence of Frequent Attendance 3045 Frequent Attenders in 2003 2609 Enlisted in 2003, 2004 and 2005 2004 1008 FAs 2005 407 FAs = pFAs 36 1601 Non-FAs 2139 Non-pFAs WHY DO THEY KEEP COMING BACK? 436 lost to follow-up in 2004 and 2005 32 110 died moved out of practice 39 255 died moved out of practice Prevalences were calculated for that subset of morbidity in which, according to the literature, frequent attenders differ most of normal attenders: diabetes mellitus, chronic cardiovascular disease, chronic respiratory disease, feelings of anxiety, feelings of depression, addictive behaviour, other psychological/psychiatric codes, all social problems and medically unexplained physical symptoms (MUPS) 2;4 . MUPS were defined according to Robbins et al. and had to comply with the definition of the Problem List 19. We determined the total number of registered medical problems as indicator of overall morbidity for the one and three year periods. Prescribed medication The yearly number of prescriptions for each patient was calculated for the following: antibiotics, painkillers, anxiolytics, hypnotics, and antidepressants. We present the average number of prescriptions of these 5 groups of medications in non-Frequent Attenders, 1-year-Frequent Attenders and persistent Frequent Attenders. Statistical analysis SPSS 14.0 for windows was used for the statistical analysis. Differences between patients groups were analysed using X2 test. Statistical significance was set at P<0.05. After checks for errors and consistency, we assessed the potential for selection bias due to loss to follow-up and death. Box 1 provides a description of our approach. Statistical analyses were performed in Stata (version 9.2). Results 1-year-Frequent Attenders, persistent Frequent Attenders and GP-workload The number of Frequent Attenders found were as follows: 2003, 3,045 (10.6%); 2004, 2,897 (10.2%); 2005, 2,499 (9.3%). Of all Frequent Attenders in 2003, 436 were lost to follow-up because they had died (71) or moved out of the practice (365) before December 31, 2005. A multivariable analysis showed (virtually) no signs of selective loss to follow up for moving out of the practice or for death (see Box 1). Of the 2,609 Frequent Attenders in 2003 who could be followed for three years, 1,008 were also Frequent Attender in 2004, while 470 continued to be a Frequent Attender in 2004 and 2005 and were a persistent Frequent Attender according to our definition. These persistent Frequent Attenders comprised 1.6% of all enlisted patients of 15 years and older in 2003 and 15.4% of all 1-year-Frequent Attenders in 2003. (See Figure 1) Compared with 1-yearFrequent Attenders, persistent Frequent Attenders are older (see diagram 1). The percentage of patients over the age of 65 years changed from 12.5% to 15.3%, the percentage of patients in the age group 4564 years changed from 26.6% to 34% and the percentage at 15-44 years decreased from 60.9% to 50.6%. The number of yearly consultations varied substantially according to age. In 2003, the mean number of consultations CHAPTER 3 37 Table 1.Mean number of GP-consultations per age group for non-Frequent Attenders, 1-year Frequent Attenders and persistent Frequent Attenders in 2003. Non-FAs1 1yFAs2 pFAs3 15-44 1.01 6.5 8.47 45-64 1.61 8.6 10.98 65+ 2.85 12.4 14.3 All patients >15 1.4 7.8 10.22 1. Non-frequent attenders 2. 1-year frequent attenders 3. persistent frequent attenders Table 2. Morbidity of non-Frequent Attenders, 1-year Frequent Attenders and persistent Frequent Attenders: prevalence and relative difference (non-Frequent attenders 100) Non-FAs1 1yFAs2 pFAs3 19120 2609 470 Diabetes mellitus 5.5 13.7 (250) 23.2 (421) Chron. Cardiovasc. disease 13.7 23.4 (170) 37.7 (275) Chron. resp. disease 9.8 17.8 (181) 27.2 (277) (feelings of) Anxiety 1.8 4.7 (261) 9.4 (522) (feelings of) Depression 3.2 6.4 (200) 8.7 (271) Addictive behaviour 1.2 2.9 (241) 4.9 (408) MUPS 6.8 13.1 (192) 25.3 (370) Social problems 1.3 2.0 (153) 7.9 (607) Psychological/psychiatric problems 9.2 20.6 (223) 37.0 (402) Number of medical problems 1.16 2.00 (172) 3.52 (303) 1. Non-frequent attenders 2. 1-year frequent attenders 3. persistent frequent attenders 38 WHY DO THEY KEEP COMING BACK? of non-Frequent Attenders (n= 19,120), 1-year Frequent Attenders (n= 2,609) and persistent Frequent Attenders (n= 470) were 1.4, 7.8 and 10.2 respectively (see Table 1). persistent Frequent Attenders, feelings of anxiety is more prevalent than feelings of depression. In 1-year Frequent Attenders feelings of depression is more prevalent. (See Table 2) In 2003, for patients of 15 years and older, 80% of all face-to-face consultations were with 37% of the registered patients. Another 37% of patients had not visited their GP at all during that year. In 2003, the 3,045 Frequent Attenders (10.6%) were responsible for 39% of all face-to-face consultations; the 470 persistent Frequent Attenders (1.6%) were responsible for 8% of all consultations. Number of prescriptions Morbidity Table 2 shows the distribution of 9 medical problems or diagnoses across the three categories of non- and frequent attenders. The most important findings are the substantial differences in morbidity for social and psychological/psychiatric problems and the high percentage of persistent Frequent Attenders with cardiovascular disease (37.7 %) and MUPS (25.3 %). Persistent Frequent Attenders present with more medical problems (3.52) than 1-year Frequent Attenders (2.0) and non-Frequent Attenders (1.16). Age follows the predictable pattern of the older the patient, the more consultations and the more medical problems. Compared with both other groups we see in persistent Frequent Attenders especially more social problems, more feelings of anxiety and more addictive behaviour. These persistent Frequent Attenders differ less as far as the prevalence of chronic somatic diseases is concerned. In Compared to non- and 1-year-Frequent Attenders, persistent Frequent Attenders received more prescriptions for painkillers antibiotics, antidepressants, anxiolytics and sleeping tablets. Especially the high number of prescriptions for painkillers and anxiolytics in Frequent Attenders is remarkable. (See Table 3) Discussion Main findings When analysing the consultations of all enlisted adult patients from 5 primary health centres during 3 consecutive years, we found that frequent attending is usually a self-limiting condition. One out every seven (15.4%) of patients who were a Frequent Attender in 2003 (or 18% of those FAs who were enlisted for all three years) remained a Frequent Attender during two consecutive years. These persistent Frequent Attenders make up 1.6% of all enlisted patients of 15 years and older in 2003. GPs held about seven times more consultations with persistent Frequent Attenders compared with non-Frequent Attenders. Compared with non- and 1-yearFrequent Attenders, persistent Frequent Attenders presented more social problems, more feelings of anxiety, more addictive behaviour and MUPS and they received more prescriptions for psychotropic medication. CHAPTER 3 39 Table 3. Mean number of prescriptions in non-Frequent attenders, 1-year-Frequent Attenders and persistent Frequent Attenders and relative difference (non-Frequent attenders 100) Non-FAs1 1yFAs2 pFAs3 19.120 2.609 470 Antibiotics 0.18 0.7 (388) 0.88 (488) Painkillers 0.51 2.3 (457) 2.91 (570) Anxiolytics 0.20 0.9 (450) 1.3 (650) Hypnotics 0.19 0.7 (368) 0.99 (521) Antidepressants 0.22 0.9 (409) 1.15 (523) 1. Non-frequent attenders 2. 1-year frequent attenders 3. persistent frequent attenders Strength and limitations An important strength of our study is the size and the longitudinal character of the dataset and the experience of the participating GPs. Most GPs have participated in the registration network for over 10 years and are used to accepting regular feedback on their registration activities. Prescriptions are extracted from the GPs’ Electronic Medical Record and the number of actual prescriptions is therefore reliable although the amount of prescribed drugs is not. Prescription data in general practice are generally considered to be of higher quality than data on diagnoses 20. As we used routinely collected data and did not plan any intervention in the normal practice 40 routine, our data reflects the day-to-day business of general practice. Furthermore, the demographic data are accurate. A limitation of our study, however, is that the data are restricted to “what the GP knows and registered”. In particular, the problem lists could be inflated (if resolved problems are not removed) or subject to underreporting. Underreporting could be the case for patients with a low consultation frequency – thus inflating the contrast between Frequent Attenders en non-Frequent Attenders – and for patients who are relatively new in the practice. As the problem lists of all participating GPs are subject to evaluation on a regular basis we think this problem is being dealt with as well as possible 21. These problem WHY DO THEY KEEP COMING BACK? lists therefore seem quite valid 22;23. Many patients who suffer from an incurable disease become frequent attenders in the months prior to their death. Although our results may include terminally ill patients, only few persistent frequent attenders were incurably sick and died soon after the study period (see box 1). The GP practices in this study are situated in an urban area. This means that the results cannot be generalized and compared with practices in more rural areas. Unfortunately, SocioEconomic-level and ethnicity were not registered. Relevant literature There is substantial literature about the characteristics and morbidity of 1-year Frequent Attenders. The few longitudinal studies show regression of attendance rates to the mean in the longer run 15;15;16;24;25. However, studies on persistent frequent attendance used different definitions of Frequent Attendance and lacked the power to detect differences in morbidity and prescriptions. Several trials have been conducted to test interventions for changing consultation behaviour and/ or morbidity of Frequent Attenders 12. Only one study consisting of 2 RCTs used frequent attendance over a period of 2 years 13;14. All others included 1-yearFrequent Attenders 26-28. Although no study found evidence to support the possibility that healthcare utilization of Frequent Attenders can be influenced, the study that included Frequent Attenders for two years did find evidence that treatment of major depressive disorder in a subgroup of depressed Frequent Attenders improved the patients’ symptoms and quality of life. Implications for future research or clinical practice. Knowing that frequent attendance is predominantly a temporary phenomenon and because of the continuous high workload, the high prevalence of diseases and the considerable use of medication, we think that only persistent Frequent Attenders deserve further attention. Regarding the important role of psychological and psychiatric problems (especially anxiety) and social problems in persisting frequent attendance it seems logical to focus on these problems in Frequent Attenders in order to try to improve their quality of life and to prevent the continuation of frequent consulting behaviour. Conclusion We conclude that, compared with normal attenders, 1-year-Frequent Attenders have many somatic and psychiatric problems, are prescribed much medication, including psychotropic medication, and that they constitute a substantial part of the clinical work of a GP. One out of every seven 1-year Frequent Attenders persists to consult frequently during a period of two consecutive years. Compared to 1-year Frequent Attenders, persistent Frequent Attenders have even more consultations with their GP, suffer from more morbidity (especially social, psychiatric and MUPS) and are prescribed more medication (especially psychotropic medication). CHAPTER 3 41 References (1) Neal RD, Heywood PL, Morley S, Clayden AD, Dowell AC. Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br J Gen Pract 1998; 48(426):895-898. (2) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (3) Smits FT, Mohrs J, Beem E, Bindels PJ, van Weert HC. Defining frequent attendance in general practice. BMC Fam Pract 2008; 9(1):21. (4) Gill D, Sharpe M. Frequent consulters in general practice: a systematic review of studies of prevalence, associations and outcome. J Psychosom Res 1999; 47(2):115-130. (5) de Waal MW, Arnold IA, Eekhof JA, Assendelft WJ, van Hemert AM. Followup study on health care use of patients with somatoform, anxiety and depressive disorders in primary care. BMC Fam Pract 2008; 9(1):5. (6) Little P, Somerville J, Williamson I, Warner G, Moore M, Wiles R et al. Psychosocial, lifestyle, and health status variables in predicting high attendance among adults. Br J Gen Pract 2001; 51(473):987-994. (10) Hansagi H, Olsson M, Sjoberg S, Tomson Y, Goransson S. Frequent use of the hospital emergency department is indicative of high use of other health care services. Ann Emerg Med 2001; 37(6):561-567. (11) Reid S, Wessely S, Crayford T, Hotopf M. Frequent attenders with medically unexplained symptoms: Service use and costs in secondary care. British Journal of Psychiatry 2002; 180(3):248-253. (12) Smits FTM, Wittkampf KA, Schene A, Bindels PJE, van Weert HCP. Interventions on frequent attenders in primary care. A systematic literature review. Scand J Prim Health Care 2008; 26(2):111. (13) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (14) Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ, Helstad CS. Costeffectiveness of systematic depression treatment for high utilizers of general medical care. Arch Gen Psychiatry 2001; 58(2):181-187. (15) Ward AM, Underwood P, Fatovich B, Wood A. Stability of attendance in general practice. Fam Pract 1994; 11(4):431-437. (7) Verhaak PF, Meijer SA, Visser AP, Wolters G. Persistent presentation of medically unexplained symptoms in general practice. Fam Pract 2006; 23(4):414-420. (16) Botica MV, Kovacic L, Tiljak MK, Katic M, Botica I, Rapic M et al. Frequent attenders in family practice in Croatia: Retrospective study. Croatian Medical Journal 2004; 45(5):620-624. (8) Booth BM, Ludke RL, Wakefield DS, Kern DC, du Mond CE. Relationship between inappropriate admissions and days of care: implications for utilization management. Hosp Health Serv Adm 1991; 36(3):421-437. (17) Carney TA, Guy S, Jeffrey G. Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 2001; 51(468):567-569. (9) Brandon WR, Chambers R. Reducing emergency department visits among high-using patients. J Fam Pract 2003; 52(8):637-640. 42 (18) Lamberts H, Wood M e. International classification of primary care. Oxford: Oxford University Press; 1988. (19) Robbins JM, Kirmayer LJ, Hemami S. Latent variable models of functional somatic distress. J Nerv Ment Dis 1997; 185(10):606-615. WHY DO THEY KEEP COMING BACK? (20) Thiru K, Hassey A, Sullivan F. Systematic review of scope and quality of electronic patient record data in primary care. BMJ 2003; 326(7398):1070. (21) De LS, Stephens PN, Adal N, Majeed A. Does feedback improve the quality of computerized medical records in primary care? J Am Med Inform Assoc 2002; 9(4):395-401. (22) Brouwer HJ, Bindels PJ, van Weert HC. Data quality improvement in general practice. Fam Pract 2006; 23(5):529536. (23) Jordan K, Porcheret M, Croft P. Quality of morbidity coding in general practice computerized medical records: a systematic review. Fam Pract 2004; 21(4):396-412. (24) Carney TA, Guy S, Jeffrey G. Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 2001; 51(468):567-569. (25) Andersson S-O, Lynoe N, Hallgren C-G, Nilsson M. Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scandinavian Journal of Primary Health Care 2004; 22(2):91-94. (26) Christensen MB, Christensen B, Mortensen JT, Olesen F. Intervention among frequent attenders of the outof-hours service: a stratified cluster randomized controlled trial. Scand J Prim Health Care 2004; 22(3):180-186. (27) Katon W, von Korff M, Lin E, Bush T. A randomized trial of psychiatric consultation with distressed high utilizers. General Hospital Psychiatry /3; 14(2):86-98Record. (28) Olbrisch ME. Evaluation of a stress management program for high utilizers of a prepaid university health service. Med Care 1981; 19(2):153-159. (29) Hernan MA, Hernandez-Diaz S, Robins JM. A structural approach to selection bias. Epidemiology 2004; 15(5):615-625. CHAPTER 3 43 chapter 4 PREDICTABILITY OF PERSISTENT FREQUENT ATTENDANCE: A HISTORIC 3-YEAR COHORT STUDY Frans T. Smits, Henk J. Brouwer, Henk C. van Weert, Aart H. Schene, Gerben ter Riet Br. J. Gen. Pract. 2009 Feb; 59(559): e44-50 ABSTRACT Background Few patients who attend frequently continue to do so. While transient frequent attendance may be readily explicable, persistent frequent attendance often is not. Besides chronic morbidity persistent frequent attenders may have hidden illness. They increase GPs’ workload while reducing work satisfaction. It is neither reasonable, nor efficient to target diagnostic assessment and intervention at transient frequent attenders. Aim To develop a prediction rule for selecting persistent frequent attenders using readily available information from GPs’ electronic medical records. Design A historic 3-year cohort study Method We used data on 28,860 adult patients from 2003 to 2005. Frequent attenders were patients whose attendance rate ranked in the (age and sex adjusted) top 10 percent during 1 year (1-year frequent attenders) or 3 years (persistent frequent attenders). Using bootstrapped multivariable logistic regression analysis, we determined which predictors contained information on persistent frequent attendance. Results Out of 3045 1-year frequent attenders 470 (15.4%) became persistent frequent attender. The prediction rule could update this prior probability to 3.3% (lowest value) or 43.3% (highest value). However, the 10th and 90th centile of the posterior probability distribution were 7.4% and 26.3%, respectively, indicating that the model performs modestly. The area under the receiver operating characteristics curve was 0.67 (95% confidence limits 0.64 and 0.69). 46 WHY DO THEY KEEP COMING BACK? Conclusions Among 1-year frequent attenders, six out of seven are transient frequent attenders. With the present indicators our rule performs modestly in selecting those at risk of becoming persistent frequent attender. More information or complementary diagnostic tests seem needed to construct a rule with sufficient performance for efficient risk stratification in clinical trials. CHAPTER 4 47 Introduction It is estimated that about 80% of a GP’s clinical work is spent on 20% of his/ her patients, and that one in every seven consultations is with patients who rank in the top 3% of the attendance rate.1 Frequent attendance is often defined as an age and sex-adjusted attendance rate ranking in the top 10 centile within a time frame of one year.2;3 Although longitudinal studies on frequent attenders are scarce, we know that most frequent attenders frequently attend their GP for a short period of time only.4-7 It is neither reasonable, nor efficient to target extensive diagnostic assessment, monitoring, and intervention at transient 1-year frequent attenders Trials on the effect of (mainly psychiatric) interventions on morbidity and attendance rates showed conflicting results.8 No study showed convincing evidence that an intervention improves quality of life or morbidity of frequent attending primary care patients, although an effect might exist in a subgroup of depressed frequent attenders.9-11 For this subgroup one trial concluded that, in the year following the intervention, patients in the intervention Box 1. Approach to the multivariable analysis Loss to follow-up 368 patients (12%) were lost at some point over the two years of follow-up. We argued that, in theory, a potential frequent attender might move out of the practice due to dissatisfaction with care. The resulting selection bias may attenuate associations found between the selected indicators and frequent attendance. We tested our hypothesis in a multivariable logistic regression analysis with an indicator variable “1 = moved house” and “0 otherwise” as the dependent variable and 9 independent indicators (see below). Our hypothesis was not confirmed. On the contrary, we found some evidence that those with at least one chronic somatic illness were less likely to have moved out of the practice(odds ratio 0.73 (95%CI from 0.54 to 0.99)); all other associations were neither strong nor significant. These results support the view that important selection bias is unlikely. Sixty-eight patients (2.2%) had died over the two year follow-up period, but since, by definition, these patients cannot become 3-year frequent attenders, selection bias by death is impossible. Variable selection Frequent attendance during all three years, coded as 1, and zero otherwise was the dependent variable. Independent variables: Continuous variables (age and the number of problems on the GP’s problem list) were assessed for linear association with the dependent variable using a graphical method proposed by Harrell to avoid model mis-specification1. Presence of diabetes mellitus and/or chronic respiratory illness and/or chronic cardiovascular illness was coded as 1, absence of any of the above as zero (52 had all three, 316 had two, 891 one, 1786 none. 48 WHY DO THEY KEEP COMING BACK? Similarly, presence of psychological and/or social problems including (feelings of) anxiety, (feelings of) depression, and/or substance abuse were combined (0 had all five, 1 had four, 33 three, 371 two, 285 one, and 2355 none). The use of antidepressants, anxiolytics, and/or hypnotics was similarly combined (118 patients used all three types of drugs, 290 two, 408 one, and 2107 none). Thus, the 9 candidate predictors, modelled as 11 variables, included: 1. age at baseline (continuous), 2. sex, 3. number of problems on the problem list (continuous), 4. any of the three chronic somatic illnesses just mentioned (yes/no), 5. any psychological/social problem (yes/no), 6. any medically unexplained physical problem (yes/no), 7. psychoactive medication (yes/no), 8. average monthly number of prescriptions for antibiotics (0 = reference category; 1-2; >2), 9. average monthly number of prescriptions for analgesics (0 = reference category; 1-4; >4). A final model was selected using bootstrapped forward stepwise logistic regression analysis which was performed 100 times2. The p-values for entry of variables into and removal from the model were 0.10 and 0.15, respectively. Candidate predictors had to be selected 70 times or more to be eligible for the final model. The final model’s fit was tested using the Hosmer-Lemeshow test (10 groups) and accounted for intracluster correlation within general practices by using robust variance estimation according to Huber and White3. Adding interaction terms to the final model, we assessed subgroup effects in the following subgroups requiring a p-value < 0.10 for significance: coexistence of a documented somatic and psychosocial problem; coexistence of a psychosocial problem and prescription of pain medication; female sex and prescription of pain medication. The regression coefficients of the final model were used to compute the probabilities of being a three year FA. The final model’s area under the receiver operating characteristics (AUCROC) curve was calculated as a summary of predictive power. The final model was fitted 500 times using bootstrap methodology and the corresponding ROC curves were used to construct a more robust confidence interval (CI) around the area under the curve thus counteracting the influence of observations unique to our data set. Reference List 1. Harrell F. Regression Modeling Strategies. 2001. New York, Springer. Ref Type: Generic 2. Sauerbrei W, Schumacher M. A bootstrap resampling procedure for model building: application to the Cox regression model. Stat Med 1992; 11(16):2093-2109. 3. Williams RL. A note on robust variance estimation for cluster-correlated data. Biometrics 2000; 56(2):645-646. CHAPTER 4 49 Figure 1. Flow diagram: Persistence of Frequent Attendance 3045 Frequent Attenders in 2003 436 lost to follow-up in 2004 and 2005 2609 Enlisted in 2003, 2004 and 2005 2004 1008 FAs 2005 407 FAs = pFAs 1601 Non-FAs 2139 Non-pFAs group had, on average, 47 depression-free days more (5% CI from 27 to 68).11 There is no evidence that it is possible to influence health care utilization of frequent attenders. All trials except one included patients that attended frequently during one year.12 Using information readily available in GPs’ electronic medical records, we set out to develop a prediction rule to help GPs to identify, among 1-year frequent attenders, those at extremely low or high risk of becoming persistent frequent attender. Such a rule, in addition to being clinically useful, may also support the selection of more homogeneous patient groups in future randomized trials among (subgroups of) persistent frequent attenders. 50 32 110 died moved out of practice 39 255 died moved out of practice Methods Patient population Five primary health care centres in Amsterdam provided data for this study. These centres participate in the GP-based continuous morbidity registration network of the Department of General Practice, Academic Medical Centre - University of Amsterdam. In this network EMR data are extracted for research purposes. The studied patients have a lower socioeconomic level, are of more non-western descent and are slightly younger than the Dutch population. The participating GPs use a problem-oriented registration method. For this study we used the numbers of face-to-face consultations with the GPs, the lists of current medical problems as registered and coded by the WHY DO THEY KEEP COMING BACK? GP using the International Classification of Primary Care (ICPC) and the number of a selection of prescriptions of all patients from 1 January 2003 through 31 December 2005. Selection of one-year frequent attenders and persistent frequent attenders Frequent attenders were defined as those patients whose attendance rate ranked nearest to the top 10th centile of their sex and age group (15-30; 31-45; 46-60; 61+) .2;3 Frequent attenders were determined for each of the years 2003, 2004 and 2005. We took as a starting point the selected frequent attenders of 2003. We defined persistent frequent attenders as those patients who were both registered and a frequent attender during all three years. Only face-to-face consultations with GPs (consultations in the office and house calls) were included. Consultations with other practice staff were excluded because these contacts are mostly initiated by the GP or his/her staff and are related to controlling chronic diseases. We determined the mean number of consultations per age and sex group for frequent attenders and non-frequent attenders. Patients younger than 15 years were excluded, because their consultations often depend on their parents. Definition and extraction of predictor information In the problem-oriented approach to medical record keeping a patient may have a list of current medical problems, also called problem list. Different from the definition used in the UK, in the Netherlands a current medical problem is defined by the GP as: 1. Any medical problem (disease or complaint) which needs continuing medical attention or monitoring. 2. Any complaint or disease present for more than 6 months (excluding all (minor) short episodes). Every problem on this list was coded by the GPs using the International Classification of Primary Care.13 Problem lists were extracted at the end of 2003 and 2005. The prevalence of each medical problem was calculated for 1-year frequent attenders at the end of the first year, for persistent frequent attenders at the end of the third year. From the electronic medical record we extracted those prescriptions and medical problems in which, according to the literature, frequent attenders and non-frequent attenders differed most: Number of prescriptions (for analgesics, tranquilizers, antidepressants and antibiotics), diabetes mellitus, chronic cardiovascular disease, chronic respiratory disease, (feelings of) anxiety, (feelings of) depression, addictive behaviour, any psychological/psychiatric problem, all social problems and medically unexplained physical symptoms (MUPS). 3;14 MUPS were defined according to Robbins et al and complied with the definition of the Problem List. 15 (See appendix 1 for the used ICPC-codes) Statistical analysis We applied a multivariable analysis using all above-mentioned information CHAPTER 4 51 Table 1. Univariate associations of candidate predictors with persistent frequent attendance (pFA), the dependent variable*. Predictor (crude) Odds ratio 95% confidence interval limits Age¶ 1.01 1.00 – 1.017 Sex, female 1.46 1.14 – 1.87 Number of active problems¶ 1.21 1.16 – 1.25 Any chronic somatic illness 1.97 1.67 – 2.33 Any psychological problem 2.18 1.73 – 2.76 Medically unexplained complaint 2.02 1.55 – 2.62 Any psychoactive medication 1.50 1.21 – 1.86 Mean montly number of analgesic prescriptions: 0 1 Reference category 1-4 1.83 1.48 – 2.25 >4 2.56 1.98 – 3.30 1 Reference category 1-2 1.21 0.99 – 1.48 >2 1.46 0.98 – 2.18 Mean monthly number of antibiotic prescriptions: 0 * ¶ Based on 3045 observations; 470 pFAs (dependent variable = 1); modelled as a continuous variable; All other variables were modelled as dummies. as predictors for persistence of frequent attendance (See box 1). After checks for errors and consistency we assessed the potential for selection bias due to loss to follow-up and death and used bootstrapped stepwise logistic regression to select the variables for the final model. Box 1 provides a detailed description of our analytical approach. Statistical analyses were performed in Stata (version 9.2). 52 Results (Persistent) frequent attenders Of the 2609 frequent attenders in 2003 who could be followed for three years, 1008 (38.6%) also frequently attended in 2004, while 470 (18.0%) continued to do so in 2004 and 2005 and were persistent frequent attender according to our definition (See figure 1). These persistent frequent attenders comprised 1.6% of all registered patients of 15 years and older in 2003. We studied selection bias, but found (virtually) none for moving out of practice or for death (see box 1). WHY DO THEY KEEP COMING BACK? Table 2. Associations between the five predictors retained in the final model and persistent frequent attendance (pFA), the dependent variable.* * ¶ Predictor (adjusted) Odds ratio 95% confidence interval limits Age ¶ 0.99 0.98 – 1.00 Number of active problems ¶ 1.13 1.05 – 1.22 Any chronic somatic illness 1.55 1.25 – 1.93 Any psychological problem 1.72 1.30 – 2.27 Mean montly number of analgesic prescriptions: 0 1 Reference 1-4 1.77 1.41 – 2.23 >4 2.06 1.59 – 2.66 Based on 3045 observations; 470 pFAs (dependent variable = 1); modelled as a continuous variable; All other variables were modelled as dummies. Prediction of persistent frequent attendance Table 1 shows the univariate associations of all candidate predictors with the dependent variable, persistent frequent attendance. Five predictors were retained in the final model: age, the number of problems on the GP’s problem list, presence of any of three chronic somatic illnesses (diabetes mellitus, cardiovascular illness, and respiratory illness), presence of a psychological/social problem, and the use of pain medication (Table 2). None of the interaction effects proved significant at the 10% level. The prior probability of 15.4% (470/3045) of persistent frequent attendance could be updated, using the model, to at best 3.3 % (lowest value) or 43.3% (highest value). The 10th and 90th centile of the posterior probability distribution were 7.4% and 26.3%, respectively, indicating that the model performs neither very good to rule out persistent frequent attendance nor to rule it in. The Hosmer-Lemeshow test showed a p-value of 0.254, thus indicating no strong evidence against good model fit. As a summary of the model’s overall discrimination, the AUCROC was 0.67 (bootstrapped bias corrected 95%CI from 0.64 to 0.69). Discussion Summary of main findings In a historic 3-year cohort study, we found that 15.4 percent of all 1-year frequent attenders persisted in this behavior during two consecutive years. Persistent frequent attenders constituted less than 2% of all registered patients 15+ of age. It proved difficult to predict which 1-year frequent attender persists in frequent consulting behaviour using present readily available information from GPs’ electronic medical records. CHAPTER 4 53 Strength and limitations of this study Comparison with existing literature An important strength of our study is the size and the longitudinal character of the dataset and the experience of the participating GPs in recording and coding the problem lists. Most GPs have participated in the registration network for over 10 years and are used to feedback on their registration activity. The problem lists have been monitored over the years and differences between doctors have been regularly discussed.16 Prescriptions There is substantial literature about the characteristics and morbidity of frequent attenders.3;14 It is striking that almost are extracted from the electronic medical record and reflect the number of actual prescriptions. Prescription data in general practice may be generally considered to be of higher quality than diagnosis-oriented data.17 Our study was based on routinely collected data reflecting everyday general practice in The Netherlands. As far as we know our study is the first predicting persistence of frequent attendance with information readily available to GPs. Routine data that are readily available have their limitations. For example, problem lists may be inflated (by not removing resolved problems) or subject to underreporting. Moving out of practice was a reason for exclusion, as followup of these patients was not possible. Unfortunately, ethnicity and socio economic-level are not (sufficiently) registered in the current electronic medical record. This precluded an analysis of the interaction between ethnicity and several other predictors to explore the role of ethnicity in more detail. 54 all descriptive literature about frequent attendance is produced in countries with some kind of list system: the United Kingdom, the Scandinavian countries, and Health Maintenance Organizations in the US.3;14 Most research on frequent attenders however is cross-sectional and uses oneyear attendance rates. In particular, 1-year frequent attenders have been reported to use more analgesics, more antibiotics and more tranquilizers.18;19 High attendance rates are also found for patients with medically unexplained somatic symptoms, health anxiety and perceived poor health.20-22 The few longitudinal studies show attendance rates to regress to the mean in the longer run, with only 20-30% of frequent attenders continuing to attend frequently in the following year. 4-7 These studies on persistent frequent attendance however use different definitions of frequent attenders and lack the power to detect factors associated with transient frequent attendance becoming persistent. In one study psychological distress, as measured with two psychometric scales, was found to increase the risk of future daytime frequent attendance of adult patients in family practice.23 As frequent attendance proves to be mostly a transient problem, interventions in 1-year frequent attenders do not seem worthwhile. Several trials have been conducted to test interventions to change consultation behaviour and/or morbidity of frequent attenders.8 Only one study used frequent WHY DO THEY KEEP COMING BACK? attendance during 2 years.10;11 All others included 1-year frequent attenders.24-26 Although none of the studies found evidence that it is possible to influence health care utilization by frequent attenders, the one that included frequent attenders during two years did find evidence that treatment of major depressive disorder in a subgroup of depressed frequent attenders improved their symptoms and quality of life.10;11 Implications for future research or clinical practice From the viewpoint of delivering good care GPs do not have the reason or the instruments to look for unmet health-care needs among one year frequent attenders. Both psychological and somatic chronic diseases and complaints predispose modestly a one-year frequent attender to become a persistent frequent attender. However, as the predictive power (for inclusion as well as for exclusion) of our rule proved to be small there might be other reasons for persistence of frequent attendance. Because our study was not a prospective cohort-study, we cannot exclude the existence of ‘hidden morbidity’ among persistent frequent attenders. Further studies are needed to decide, first, whether there exist undiscovered morbidity among persistent frequent attenders and, secondly, whether it is possible and worthwhile to construct a rule with sufficient performance for risk stratification by using more information about the patient or from diagnostic tests. Conclusion Among 1-year frequent attenders, about six out of seven are transient frequent attenders. Information from GPs’ electronic medical records may be used to identify those at low and higher risk of becoming persistent frequent attender. With the present indicators, available in the electronic medical record, our rule performs modestly in selecting those at risk of becoming persistent frequent attender. Ethics committee According to the Medical Research Involving Human Subjects Act (WMO), formal approval for this research project by a Medical Ethics Committee was not necessary. The academic GP network extracts data according to strict guidelines for the privacy protection of patients and GPs. In addition we sought and obtained permission for this work from the board of the network Competing interests None. Acknowledgments We thank the GPs involved in the Network of General Practitioners of the Academic Medical Centre/University of Amsterdam (HAG-net-AMC) for their continuous efforts to keep the electronic medical records updated. CHAPTER 4 55 References (1) Neal RD, Heywood PL, Morley S, Clayden AD, Dowell AC. Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br J Gen Pract 1998; 48(426):895-898. (2) Smits FT, Mohrs JJ, Beem EE, Bindels PJ, van Weert HC. Defining frequent attendance in general practice. BMC Fam Pract 2008; 9(1):21. (3) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (4) Ward AM, Underwood P, Fatovich B, Wood A. Stability of attendance in general practice. Fam Pract 1994; 11(4):431-437. (5) Botica MV, Kovacic L, Tiljak MK, Katic M, Botica I, Rapic M et al. Frequent attenders in family practice in Croatia: Retrospective study. Croatian Medical Journal 2004; 45(5):620-624. (6) Carney TA, Guy S, Jeffrey G. Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 2001; 51(468):567-569. pattern at the family practitioner. Scandinavian Journal of Primary Health Care 2004; 22(2):91-94. (7) Andersson S-O, Lynoe N, Hallgren C-G, Nilsson M. Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendanceUniversity Press; 1988. (8) Smits FT, Wittkampf KA, Schene AH, Bindels PJ, van Weert HC. Interventions on frequent attenders in primary care. A systematic literature review. Scand J Prim Health Care 2008; 26(2):111-116. (9) Schreuders B, van MH, Smit J, Rijmen F, Stalman W, van OP. Primary care patients with mental health problems: outcome of a randomised clinical trial. Br J Gen Pract 2007; 57(544):886-891. 56 (10) Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ, Helstad CS. Costeffectiveness of systematic depression treatment for high utilizers of general medical care. Arch Gen Psychiatry 2001; 58(2):181-187. (11) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (12) Smits FTM, Wittkampf KA, Schene A, Bindels PJE, van Weert HCP. Interventions on frequent attenders in primary care. A systematic literature review. Scand J Prim Health Care 2008; 26(2):111. (13) Lamberts H, Wood M e. International classification of primary care. Oxford: Oxford (14) Gill D, Sharpe M. Frequent consulters in general practice: a systematic review of studies of prevalence, associations and outcome. J Psychosom Res 1999; 47(2):115-130. (15) Robbins JM, Kirmayer LJ, Hemami S. Latent variable models of functional somatic distress. J Nerv Ment Dis 1997; 185(10):606-615. (16) Brouwer HJ, Bindels PJ, Weert HC. Data quality improvement in general practice. Fam Pract 2006; 23(5):529536. (17) Thiru K, Hassey A, Sullivan F. Systematic review of scope and quality of electronic patient record data in primary care. BMJ 2003; 326(7398):1070. (18) Bergh H, Marklund B. Characteristics of frequent attenders in different age and sex groups in primary health care. Scand J Prim Health Care 2003; 21(3):171-177. (19) Vedsted P, Sorensen HT, Mortensen JT. Drug prescription for adult frequent attenders in Danish general practice: a population-based study. Pharmacoepidemiol Drug Saf 2004; 13(10):717-724. WHY DO THEY KEEP COMING BACK? (20) Little P, Somerville J, Williamson I, Warner G, Moore M, Wiles R et al. Psychosocial, lifestyle, and health status variables in predicting high attendance among adults. Br J Gen Pract 2001; 51(473):987-994 (30) Williams RL. A note on robust variance estimation for cluster-correlated data. Biometrics 2000; 56(2):645-646. (21) Verhaak PF, Meijer SA, Visser AP, Wolters G. Persistent presentation of medically unexplained symptoms in general practice. Fam Pract 2006; 23(4):414-420. (22) de Waal MW, Arnold IA, Eekhof JA, Assendelft WJ, van Hemert AM. Followup study on health care use of patients with somatoform, anxiety and depressive disorders in primary care. BMC Fam Pract 2008; 9(1):5. (23) Vedsted P, Fink P, Olesen F, MunkJorgensen P. Psychological distress as a predictor of frequent attendance in family practice: a cohort study. Psychosomatics 2001; 42(5):416-422. (24) Christensen MB, Christensen B, Mortensen JT, Olesen F. Intervention among frequent attenders of the outof-hours service: a stratified cluster randomized controlled trial. Scand J Prim Health Care 2004; 22(3):180-186. (25) Katon W, Von KM, Lin E, Bush T, Russo J, Lipscomb P et al. A randomized trial of psychiatric consultation with distressed high utilizers. Gen Hosp Psychiatry 1992; 14(2):86-98. (26) Olbrisch ME. Evaluation of a stress management program for high utilizers of a prepaid university health service. Med Care 1981; 19(2):153-159. (27) Hernan MA, Hernandez-Diaz S, Robins JM. A structural approach to selection bias. Epidemiology 2004; 15(5):615-625. (28) Harrell F. Regression Modeling Strategies. 2001. New York, Springer. Ref Type: Generic (29) Sauerbrei W, Schumacher M. A bootstrap resampling procedure for model building: application to the Cox regression model. Stat Med 1992; 11(16):2093-2109. CHAPTER 4 57 chapter 5 PREDICTABILITY OF PERSISTENT FREQUENT ATTENDANCE IN PRIMARY CARE: A TEMPORAL AND GEOGRAPHICAL VALIDATION STUDY. Frans T. Smits, Henk J. Brouwer, Koos H. Zwinderman , Marjan van den Akker, Ben van Steenkiste, Jacob Mohrs, Aart H. Schene, Henk C. van Weert, Gerben ter Riet PLoS One. 2013 Sep 5;8(9):e73125. ABSTRACT Background Frequent attenders are patients who visit their general practitioner exceptionally frequently. Frequent attendance is usually transitory, but some frequent attenders become persistent. Clinically, prediction of persistent frequent attendance is useful to target treatment at underlying diseases or problems. Scientifically it is useful for the selection of highrisk populations for trials. We previously developed a model to predict which frequent attenders become persistent. Aim To validate an existing prediction model for persistent frequent attendance that uses information solely from General Practitioners’ electronic medical records. Method We applied the existing model (N=3,045, 2003-2005) to a later time frame (2009-2011) in the original derivation network (N=4,032, temporal validation) and to patients of another network (SMILE; 2007-2009, N=5,462, temporal and geographical validation). Model improvement was studied by adding three new predictors (presence of medically unexplained problems, prescriptions of psychoactive drugs and antibiotics). Finally, we derived a model on the three data sets combined (N=12,539). We expressed discrimination using histograms of the predicted values and the concordancestatistic (c-statistic) and calibration using the calibration slope (1=ideal) and Hosmer-Lemeshow tests. Results The existing model (c-statistic 0.67) discriminated moderately with predicted values between 7.5 and 50 percent and c-statistics of 0.62 and 0.63, for validation in the original network and SMILE network, respectively. Calibration (0.99 originally) was better in SMILE than in the original network (slopes 0.84 and 0.65, respectively). Adding information on the three new predictors did not importantly improve the model (c-statistics 0.64 and 0.63, respectively). Performance of the model based on the combined data was similar (c-statistic 0.65). 60 WHY DO THEY KEEP COMING BACK? Conclusion This external validation study showed that persistent frequent attenders can be prospectively identified moderately well using data solely from patients’ electronic medical records. CHAPTER 5 61 Introduction Some patients visit their general practitioner (GP) relatively often. This frequent attendance is mostly defined as an age- and sex-adjusted attendance rate ranking in the top 10% within a time frame of 1 year.1;2 Frequent attenders (FAs) are responsible for 39% of all face-to-face consultations of their GPs and persistent frequent attenders (those 1.6 percent who frequently attend during three consecutive years or more; pFA) are responsible for about 8% of face-to-face consultations.3 Frequent attenders and, in particular, persistent frequent attenders have relatively many somatic, psychiatric and social problems.3 Although longitudinal studies on frequent attenders are scarce, it is known that most frequent attenders frequently attend their GPs for a short period of time only.3-7 It seems neither reasonable, nor efficient to target extensive diagnostic assessment, monitoring, and intervention at transient 1-year frequent attenders. However, patients who continue to attend frequently may require special attention, and potential effective interventions should probably be targeted at this group. Prediction of persistent frequent attendance may therefore be clinically useful if effective treatment of underlying medical problem and (thereby) prevention of this persistence is available. Scientifically a prediction model for pFAs may be useful to help select more homogeneous high-risk populations for future randomized trials or support efficient subgroup analysis.8;9 62 A review on the effects of (mainly psychiatric) interventions on morbidity and attendance rates has shown conflicting results.10 One out of a total of 5 trials showed that a depression management program improved quality of life and the number of depression-free days of patients frequently attending the GP during two years.11;12 None of the included trials showed an effect on healthcare utilization of frequent attenders. All trials, except the one mentioned above, included patients that attended frequently during just one year.12 Therefore, some negative findings may have been due to strong regression to the mean and spontaneous ‘recovery’, making it difficult to detect any effects. A more recent Spanish study in 1-year FAs showed that a 15 hours’ training of GPs, which incorporated biopsychosocial, organizational, and relational approaches resulted in a reduction of attendance rates (mean number of annual contacts in the intervention group 13.1 against 19.4 in the usual care group).13 Using information that was readily available in GPs’ electronic medical records (EMR), we developed a prediction rule to help GPs identify, among 1-year frequent attenders, those at extremely low or high risk of becoming persistent frequent attenders (see table 1).14 With the indicators available in the EMR presented in our previous study, our rule was modestly effective in selecting those at risk of becoming persistent frequent attenders (AUC 0.67; CI 0.64-0.69). Since many diagnostic indices perform worse in a different population, (external) validation in a different primary care WHY DO THEY KEEP COMING BACK? Table 1. Original prediction rule14: Associations between five predictors and persistent frequent attendance (pFA), the dependent variable.* * ¶ Predictor (adjusted) Odds ratio 95% confidence interval limits Age¶ 0,99 0,98 – 1,00 Number of active problems¶ 1,13 1,05 – 1,22 Any chronic somatic illness 1,55 1,25 – 1,93 Any psychological problem 1,72 1,30 – 2,27 Mean montly number of analgesic prescriptions: 0 1 Reference 1-4 1,77 1,41 – 2,23 >4 2,06 1,59 – 2,66 Based on 3045 observations; 470 pFAs (dependent variable = 1); modeled as a continuous variable; All other variables were modeled as dummies. population is warranted before the use in clinical practice is advocated.15 In this study we temporally (another time frame) and geographically (another area) validated our previously derived prediction model for pFA-ship using information solely from GPs’ EMR and looked for opportunities to improve it with extra patient information. Because they are more prevalent in pFAs and theoretically likely to increase persistence of frequent attendance, we added 4 extra variables (sex, presence of medically unexplained symptoms, prescriptions of psychoactive drugs and antibiotics) to the original model and tested it for improvement on the 3 cohorts (including original cohort).3;16;17 We also explored building a more robust model based on the combined data of all three datasets. Methods Ethics statement The study was conducted according to the Dutch legislation on data protection (Ministry of Justice, the Netherlands). Ethics approval was provided through the Medical Ethics Review Committee of the Academic Medical Center of the University of Amsterdam (letter W 12_259#12.27.0295), stating that “the Medical Research involving human subjects Act (WMO) does not apply to this study and that an official approval of this study by our committee is not required”. Patient population To validate our prediction rule we used two primary care cohorts: 1. Temporal validation; Six primary healthcare centres in Amsterdam and Diemen provided data for the second CHAPTER 5 63 cohort. These centers participate in the GP-based continuous morbidity registration network of the Department of General Practice, Academic Medical Center - University of Amsterdam.14 This cohort was an enlarged version of our original cohort (4,032 vs. 3,045 adult patients) in a more recent time frame (20092011). See our original article for more details.14 2. Geographical and temporal validation; All primary healthcare centres of the Eindhoven Corporation of Primary Health Care Centres in Eindhoven provided data for the first cohort. These centers participate in the GPbased Study on Medical Information and Lifestyles Eindhoven (SMILE) of the Department of General Practice of Maastricht University.18 The patients studied were of average socioeconomic level, of more western descent, and slightly older than the general Dutch population. This cohort consisted of 5,462 patients who were FA in 2007 and we followed them to 2009. In both networks, EMR data are extracted for research purposes. The participating GPs use a problem-oriented registration method. This study used the numbers of face-to-face consultations with the GPs, the lists of current medical problems as registered and coded by the GPs using the International Classification of Primary Care (ICPC), and a selection of prescriptions of all patients.19 Selection of one-year frequent attenders and persistent frequent attenders In all cohorts, frequent attenders were defined as those adult patients whose attendance rates ranked nearest to the top 10th centile of their age group (15–30, 31–45, 46–60, 61 years) separate for men and women.1;2 Persistent frequent attenders were defined as those patients who were both registered and frequently attending during 3 consecutive years. Figure 1. Flow chart of the 3 databases. AMSTERDAM I AMSTERDAM II FAs1 in 2003 N= 3045 FAs in 2009 N= 4032 LFU2:436: • 71 (died) • 365 (moved house) pFAs3 (2003-2005) n= 470 64 WHY DO THEY KEEP COMING BACK? LFU: 608 • 147 (died) • 461 (moved house pFAs (2009-2011) n= 629 Only face-to-face consultations with GPs (consultations in the office and house calls) were included. Consultations with other practice staff were excluded because, in the practices involved, such consultations are mostly initiated and planned by GPs or their staff and cover mainly chronic disease programs. Definition and extraction of predictor information We considered potential predictors of persistent frequent attendance which were easily obtainable in all three cohorts (see table 2). In the problem-oriented approach to medical record keeping, a patient may have a list of current medical problems, also called a problem list. In the Netherlands, a current medical problem is defined as: • Any disease or complaint which, according to the GP, needs continuing medical attention or monitoring and/ or SMILE FAs in 2007 N= 5462 LFU: 1994 • Any disease or complaint present for more than 6 months and/or • Recurrent medical problems (more than 4 episodes per half year).20 Every problem on this list was coded by the GPs using the ICPC.19 The prevalence of each medical problem was calculated at the end of the year. Medically Unexplained Symptoms (MUS) were defined according to Robbins et al.21 See appendix 1 for a list of the selected ICPC-codes. Statistical analysis:Validation of model to predict persistent frequent attendance The prediction model, to be validated in the present analysis, has been derived in our previous study using (bootstrapped) multivariable logistic regression analysis. This model included the variables: age (Odds ratio (OR) 0.99 per year), number of active problems (OR 1.13 per additional problem), presence of any chronic somatic problems (OR 1.55), any psychological problems (OR 1.72) and the monthly 1. FAs = frequent attenders during 1 year 2. LFU= Lost to follow up 3. pFAs= persistent frequent attenders; frequent attender during 3 consecutive years 4. In the SMILE cohort patients who changed GP within the same primary care organisation were not properly registered. Unfortunately no distinction was made between moving house or death. pFAs (2007-2009) n= 1107 CHAPTER 5 65 Figure 2. 500 Amsterdam I Histograms showing the predicted values based on the model predictions for the three cohorts: Amsterdam I (the original (derivation) cohort) and the two external validation cohorts, Amsterdam II and SMILE. 400 300 200 The graphs illustrate the slight overoptimism of the original model and the shrinkage of the distribution, that is, the tails of the Amsterdam II and SMILE cohort distributions are slightly closer to the center and predicted values smaller than 7 percent or greater than 54% no longer occur on external validation. Y -axes are frequencies. 100 0 0 500 1 2 3 4 5 6 Amsterdam II 400 300 200 100 0 0 500 1 2 3 4 5 6 SMILE 400 300 200 100 0 0 66 1 2 3 4 5 6 WHY DO THEY KEEP COMING BACK? Figure 3. 4 In these Hosmer-Lemeshow calibration plots, each circle represents the observed mean probability of becoming a persistent frequent attender (pFA) within a decile of patients after all patients were ordered from lowest to highest predicted probability. Observer (proportion) Amsterdam I Hosmer Lemeshow plots: Observed versus predicted risk for persistent Frequent Attendance. 3 2 1 As usual, the Hosmer Lemeshow calibration top plot shows a good match between predicted and observed risks in the derivation cohort (Amsterdam I) as all circles are close to the diagonal of perfect calibration. 0 0 1 2 3 4 4 On external validation in the Amsterdam II cohort (middle graph), eight out of ten predicted values were higher than those observed and those in deciles 5, 8 and 10 (extreme right hand circle) in particular. On external validation in the SMILE cohort, predicted probabilities matched the observed ones well, except for the two highest deciles, 9 and 10. The small p-values are also partly caused by the large sample size so that small mismatches become statistically significant. Note that the vertical distance to the diagonal represents the mismatch between predicted and observed pF A probabilities. Amsterdam I to Amsterdam II p < 0,001 3 2 1 0 0 1 2 Predicted (proportion) Observer (proportion) 4 3 4 3 4 Amsterdam I to SMILE p = 0,003 3 2 1 0 0 1 2 Predicted (proportion) CHAPTER 5 67 Table 2. Comparison of the three databases A’dam I # A’dam II SMILE Time period 2003-2005 2009-2011 2007-2009 Patients n 28,680 40,320 54,620 Frequent attenders n 3,045 4,032 5,462 Number of Frequent Attenders n(%) 470 (15%) 629 (16%) 1,107 (20%) Lost to follow up’ n(%) 436 (14.3) 608 (15.1) 199 (3.6) Mean age (SD) 42.6 (18.2) 47.9 (18.5) 45.9 (18.8) 1,566 (51) 2,179 (54) 2,640 (48) 10.2 11.8 7.7 Active problems (Frequent Attenders),n(SD) 2.03 (2.16) 2.68 (2.70) 1.70 (1.55) Any chronic somatic illness n(%) 1,259(41) 1,906(47) 2,768 (50) Any psychological or social problem n(%) 690 (23) 1,028 (26) 2,781 (51) Any Medically Unexplained Symptoms n(%) 391 (13) 610 (15) 98 (2) Mean monthly number of analgesic prescriptions: 0 1,484(49) 1,889 (49) 2,759 (51) 1-4 1,061 (35) 1,446 (36) >4 500 (16) 597 (15) 2,703(50) Any psychoactive medication n (%) 938 (31) 1,230 (31) 1,775 (33) Mean monthly number of antibiotic prescriptions: 0 1,976 (65) 2,374 (59) 3,120 (57) 1-2 814 (27) 1,172 (29) >2 255 (8) 486 (12) Females n(%) * Consultations of pFAs (mean n/year) Problems on the problem list n SD pFAs # * 68 2,342 (43) indicates number indicates standard deviation indicates persistent Frequent attenders, frequent attenders during 3 years Respectively in 2005 (A’dam I), 2011 (A’dam II) and 2009 (SMILE). A’dam indicates the Amsterdam I cohort Respectively in 2005 (A’dam I), 2011 (A’dam II) and 2009 (SMILE). WHY DO THEY KEEP COMING BACK? number of analgesic prescriptions (> 4: OR 2.06).14 See Table 1. We first validated the original prediction model through the assessment of its discrimination (predictive values, c-statistic and corresponding 95% confidence intervals) and calibration (slope and corresponding 95% confidence intervals, and Hosmer Lemeshow plots and tests comparing predicted versus observed risk). A calibration slope of 1 indicates that predicted probabilities match observed risks perfectly (100%), a slope <1 indicates over-prediction.22-24 We repeated this analysis for an extended model with four extra variables. Recalculation of the prediction model in the combined cohorts Recalculation of the original model was performed using all pooled subjects from the three cohorts (update cohort, n= 12,539). Regression coefficients were obtained using logistic regression with persistent frequent attendance as the dependent variable and age, number of active problems, any chronic somatic problem, any psychological problem and the monthly number of analgesic prescriptions as predictors. Analyses were performed using SPSS for windows, version 20. Results Prediction of persistent frequent attenders Table 2 and figure 1 show the general characteristics of the three cohorts. The persistent frequent attenders comprised 15-20% of all registered adult patients in all cohorts. Compared with both Amsterdam cohorts, patients in the SMILE cohort were relatively more often pFAs, had less loss to follow-up, fewer active problems, more psychological problems and fewer medically unexplained symptoms. In SMILE the mean number of prescriptions for analgesics and antibiotics was higher, but lower for psychoactive medication. In the SMILE cohort patients who changed GP within the same primary care organization were not registered as having moved house. Unfortunately, no distinction was made between moving house or death. We corrected for loss to follow (LFU) up by measuring the prognostic index with and without correction for LFU. The results did not materially change. See appendix 2. Table 3 shows the results of the original prediction rule in the Amsterdam II cohort and the SMILE cohort. Using the original regression weights with shrunken coefficients the c-statistics were 0.67 (95% confidence interval (CI) 0.64-0.69) in the original database, 0.62 (CI: 0.600.65) in the Amsterdam II cohort and 0.63 (CI: 0.61-0.65) in the SMILE cohort. Re-estimation of the regression weights did not change the results much (table 3). As expected in low-prevalence settings, negative predictive values were high, but all other indices were of moderate size. Figure 2 shows the predictive values (predicted probabilities of becoming pFA) based on the prediction model in the three cohorts. Predictive values on external validation (AMC II and SMILE) lay between 7.5 percent and 54 percent, slightly more conservative than those in the original cohort (3.3 and 59%). CHAPTER 5 69 Table 3. Discrimination and calibration on external validation of the original prognostic index to the Amsterdam II and SMILE cohorts. number of FAs/ pFAsa A’dam I A’dam II SMILE 3,045 / 470 4,032 / 629 5,462 / 1107 Using the original regression weights# C-statistics (95% CI)b 0.67 Positive predictive value Negative predictive value 0.64-0.69 0.62 0.60-0.65 0.63 0.27 0.22 0.27 0.90 0.89 0.86 0.61-0.65 Re-estimation of the regression weights C-statistics (95% CI) 0.67 0.64-0.69 0.64 0.61-0.66 0.63 Positive predictive value 0.27 0.22 0.27 Negative predictive value 0.90 0.89 0.86 0.62-0.65 Adding 3 other predictor variablesc C-statistics (95% CI) 0.67 0.65-0.70 0.65 0.62-0.67 0.65 Positive predictive value 0.26 0.23 0.26 Negative predictive value 0.90 0.89 0.88 0.656 0.07 0.83 0.63-0.66 Calibration of the original prognostic indexd Slope (SE) a 0.99 0.08 0.06 (p)FAs: (persistent) Frequent Attenders: frequently attending patients during 1 and 3 years, respectively. Concordance statistics (95% confidence interval) Any medically unexplained symptoms; any psychoactive medication; mean monthly number of prescriptions of antibiotics Ideally, the slope should be 1, which indicates perfect calibration of predicted and observed risks. Values <1 indicate overoptimism (shrinkage), that is, high risks are overestimated, while low risks are underestimated using a model with shrunken coefficients of the original model (shrinkage coefficient 0.993)Sensitivity, specificity, likelihood ratios and positive and negative predictive values were calculated at the value where their sum was maximal (Q-point of the ROC curve). b c d # 70 WHY DO THEY KEEP COMING BACK? The effect of clustering on the health center level was negligible with intraclass correlations of 0.02-0.06 and almost no change of the regression weights. See the supporting file (Table S2). Adding three predictors (any MUS, any psychoactive medication and the mean monthly number of antibiotic prescription) hardly changed the performance (c-statistics Amsterdam II 0.65; CI 0.620.67; SMILE 0.65; CI 0.63-0.66) (see table 3). The Hosmer Lemeshow plots showed modest calibration with underestimation of the probability to become pFA (see Figure 1, upper right circles which tend to fall below the line y=x). This was confirmed by the small p-values indicating large discrepancies between predicted and observed risks. (See figure 3) Updated prediction model We updated the prediction model using all pooled patients of the three cohorts (12,539 patients). Pooling the three data sets and fitting a new model did not materially lead to important improvements (c-statistic 0.65; CI: 0.63-0.66). Discussion Summary of main findings External validation in time and place of an existing prediction model for persistent frequent attendance in primary care showed that its discrimination remained stable while calibration was reduced for the higher predictions in particular. Model extension with three plausible predictors not previously included hardly improved model performance. Strength and limitations of the study Important strengths of this study are the size and the longitudinal character of the datasets and the experience of the participating GPs in recording and coding the problem lists.20 The SMILE cohort had relatively little loss to follow-up (mostly because fewer patients ‘moved house’ because of registration limitations) and more pFAs. Knowing the way GPs and practice staff cooperate in the Netherlands, replacement of GP consultations by practice staff consultation (off-utilization bias) will be very limited.25 Prescriptions are extracted from the electronic medical record and reflect the number of actual prescriptions. Prescription data in general practice may generally be considered to be of higher quality than diagnosis-oriented data.26 The higher prescription rates of analgesics and antibiotics and the lower rates for psychoactive medication are therefore difficult to understand and may reflect the different patient populations in both cities and/or different prescription habits of the local GPs. Wennberg showed that everyday clinical practice is characterized by wide geographical variations that cannot be explained by illness severity or patient preference.27;28 The present retrospective study was based on routinely collected data and therefore reflects everyday general practice in the Netherlands. Because we wanted to predict patient behavior, we only used GP-patient consultations and not planned monitoring consultations for chronic diseases with other primary care staff. CHAPTER 5 71 However, there are also some limitations. First, there are differences between the Amsterdam and SMILE cohort which may have influenced the predictive performance of our rule. In general, within a General Practice Research Network, one distinguishes four categories of factors to explain morbidity and prescription differences: “healthcare system”, “methodological characteristics of the network”, “general practitioner”, and the “patient”. These factors and sub-factors are often interrelated.29 The differences between the cohorts in Amsterdam and Eindhoven may partly be explained by methodological differences (the shorter existence of SMILE (fewer active problems) and coding agreements (fewer MUS, but more psychological codes in SMILE)), different populations of general practitioners (shorter experience in coding problems in SMILE) and patient factors (more stable population; more females in SMILE). However, socio-demographic characteristics of populations cannot explain the differences in morbidity estimations among these cohorts.30 The problem lists may suffer from overreporting (by not removing resolved problems, for instance, depression) and underreporting (for instance personality disorder). This may have diminished the predictive power of “any psychological problem”. Moving out of practice was a reason for exclusion, as follow-up of these patients was not possible. Compared with SMILE the loss to follow-up rate was higher in Amsterdam, but in the original study this did not result in selection bias.14 Finally, the presumed higher registration 72 quality in these academic networks may diminish the generalizibility of a prediction rule derived in these networks to other practices. Formal external validation of prediction models while important is still scarce. If the original prediction model had had excellent performance, one may expect worse performance on external validation. External validation usually reveals the so-called over-optimism of the original model.31 Our original model performed moderately well and its discrimination performance remained largely intact, although the predicted risks did not match the observed risks very well for the more extreme risk predictions. Using shrunken coefficients of the original model had a very limited effect and there was no sign of selective loss to follow-up. Finally, accounting for clustering within health care center had little impact. Comparison with existing literature The few longitudinal studies about frequent attendance showed that attendance rates tend to regress to the mean over time, with only 20–30% of frequent attenders continuing to attend frequently in the following year.4-6;32 However, these studies of persistent frequent attendance used different definitions of frequent attenders and lacked the power to detect factors associated with transient frequent attendance becoming persistent. Vedsted found that psychological distress, as measured with two psychometric scales, increased the risk of future daytime frequent attendance of adult patients WHY DO THEY KEEP COMING BACK? in family practice.16 Another, small, prospective study (85 primary healthcare patients of working-age) detected as risk factors for persistent frequent attendance female sex, body mass index above 30, former frequent attendance, fear of death, alcohol abstinence, low patient satisfaction, and irritable bowel syndrome.33 Smits et al showed that with the indicators currently present in Dutch electronic medical records, a rule performed modestly in selecting those more likely to become persistent frequent attenders.14 beyond multiple univariable subgroup analyses.9;35 Conclusion Prediction of persistent frequent attendance using data solely from EMRs currently available in the Netherlands may be moderately helpful in identifying those patients at high (or low) risk of becoming persistent frequent attenders. Better predictors are needed to improve prediction. Implications for clinical practice Acknowledgments After validation and updating the existing rule only predicts persistence of frequent attendance moderately. For clinical use this rule has some significance to predict which 1yFAs have more risk to become a persistent FA. We are currently prospectively following a cohort of 623 frequent attenders for three years and hope to be able to improve predictions by incorporating better and more patient-based information, such as socioeconomic status, body mass index, health anxiety/ illness behavior, depressive complaints and anxiety. This has to be weighed against increased time and costs to collect such information. We thank the GPs involved in the Network of General Practitioners of the Academic Medical Centre/University of Amsterdam (HAG-net-AMC) and of the Academic Medical Centre/University Maastricht (SMILE) for their continuous efforts to keep the electronic medical records updated and their support. Implications for future research The model may be useful to select populations for RCTs with a higher likelihood of becoming pFA. In addition, prediction models can be used in RCTs to characterize the trial arms at baseline in a multivariable way, thus enhancing the assessment of baseline comparability.9;34 Finally, subgroup analyses using the scores from prediction models may serve to move CHAPTER 5 73 References 1. Vedsted P, Christensen MB (2005) Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 119: 118-137. 11. Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP et al (2000) Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 9: 345-351. 2. Smits FT, Mohrs J, Beem E, Bindels PJ, van Weert HC (2008) Defining frequent attendance in general practice. BMC Fam Pract 9: 21. 12. Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ et al (2001) Costeffectiveness of systematic depression treatment for high utilizers of general medical care. Arch Gen Psychiatry 58: 181-187. 3. Smits FT, Brouwer HJ, ter Riet G, van Weert HC (2009) Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC Public Health 9: 36. 4. Ward AM, Underwood P, Fatovich B, Wood A (1994) Stability of attendance in general practice. Fam Pract 11: 431-437. 5. Botica MV, Kovacic L, Tiljak MK, Katic M, Botica I et al (2004) Frequent attenders in family practice in Croatia: Retrospective study. Croatian Medical Journal 45: 620-624. 6. Carney TA, Guy S, Jeffrey G (2001) Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 51: 567-569. 7. Andersson SO, Lynoe N, Hallgren CG, Nilsson M (2004) Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scand J Prim Health Care: 91-94. 8. Wagner M, Balk EM, Kent DM, Kasiske BL, Ekberg H et al (2009) Subgroup analyses in randomized controlled trials: the need for risk stratification in kidney transplantation. Am J Transplant: 22172222. 9. Kent DM, Hayward RA (2007) Limitations of applying summary results of clinical trials to individual patients: the need for risk stratification. JAMA: 12091212. 10. Smits FT, Wittkampf KA, Schene AH, Bindels PJE, Van Weert HCPM (2008) Interventions on frequent attenders in primary care. Scandinavian Journal of Primary Health Care Vol 26 (2), -116. 74 13. Bellon JA, Rodriguez-Bayon A, de Dios LJ, Torres-Gonzalez F (2008) Successful GP intervention with frequent attenders in primary care: randomised controlled trial. Br J Gen Pract 58: 324-330. 14. Smits FTM, Brouwer H.J., ter Riet G, van Weert HC (2009) Predictability of persistent frequent attendance. A historic 3-year cohort study. Br J Gen Pract 2-2009: 114-119. 15. Steyerberg EW, Vickers AJ, Cook NR, Gerds T, Gonen M et al (2010) Assessing the performance of prediction models: a framework for traditional and novel measures. Epidemiology 128-138. 16. Vedsted P, Fink P, Olesen F, MunkJorgensen P (2001) Psychological distress as a predictor of frequent attendance in family practice: a cohort study. Psychosomatics 42: 416-422. 17. Jyvasjarvi S, Joukamaa M, Vaisanen E, Larivaara P, Kivela S et al (2001) Somatizing frequent attenders in primary health care. J Psychosom Res 50: 185192. 18. van den Akker M, Spigt MG, De Raeve L, van Steenkiste B, Metsemakers JF et al (2008) The SMILE study: a study of medical information and lifestyles in Eindhoven, the rationale and contents of a large prospective dynamic cohort study. BMC Public Health: 19-20696755. 19. Lamberts H and Wood M, eds (1988) International classification of primary care. Oxford: Oxford University Press. 20. Brouwer HJ, Bindels PJ, van Weert HC (2006) Data quality improvement in general practice. Fam Pract 23: 529-536. WHY DO THEY KEEP COMING BACK? 21. Robbins JM, Kirmayer LJ, Hemami S (1997) Latent variable models of functional somatic distress. J Nerv Ment Dis 185: 606-615. 22. Hosmer, D. W., Lemeshow, S., Sturdivant, R. X. (2013) Applied logistic regression, 3 rd edition. New York: John Wiley & Sons. 23. Lemeshow S, Hosmer DW, Jr. (1982) A review of goodness of fit statistics for use in the development of logistic regression models. Am J Epidemiol: 92106. 32. Andersson S-O, Lynoe N, Hallgren C-G, Nilsson M (2004) Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scandinavian Journal of Primary Health Care 22: 91-94. 33. Koskela TH, Ryynanen OP, Soini EJ (2010) Risk factors for persistent frequent use of the primary health care services among frequent attenders: a Bayesian approach. Scand J Prim Health Care 28: 55-61. 24. Paul P, Pennell ML, Lemeshow S (2012) Standardizing the power of the HosmerLemeshow goodness of fit test in large data sets. Stat Med 2012: Jul. 34. Hayward RA, Kent DM, Vijan S, Hofer TP (2006) Multivariable risk prediction can greatly enhance the statistical power of clinical trial subgroup analysis. BMC Med Res Methodol: 18. 25. Bellon JA, Delgado-Sanchez A, de Dios LJ, Lardelli-Claret P (2007) Patient psychosocial factors and primary care consultation: a cohort study. Fam Pract 24: 562-569. 35. Kent DM, Lindenauer PK (2010) Aggregating and disaggregating patients in clinical trials and their subgroup analyses. Ann Intern Med 2010: 51-52. 26. Thiru K, Hassey A, Sullivan F (2003) Systematic review of scope and quality of electronic patient record data in primary care. BMJ 326: 1070. 27. Wennberg JE (1987) The paradox of appropriate care. JAMA 258: 2568-2569. 28. Wennberg JE (1985) Practice variations: why all the fuss? Internist 26: 6-8. 29. van den Dungen C, Hoeymans N, Gijsen R, van den Akker M, Boesten J et al (2008) What factors explain the differences in morbidity estimations among general practice registration networks in the Netherlands? A first analysis. The influence of population characteristics on variation in general practice based morbidity estimations. BMC Public Health;14: 53-62. 30. van den Dungen C, Hoeymans N, Boshuizen HC, van den Akker M, Biermans MC et al (2011) The influence of population characteristics on variation in general practice based morbidity estimations. BMC Public Health 2011: 887. 31. Altman DG, Royston P (2000) What do we mean by validating a prognostic model? Stat Med 2000: 453-473. CHAPTER 5 75 chapter 6 MORBIDITY AND DOCTOR CHARACTERISTICS ONLY PARTLY EXPLAIN THE SUBSTANTIAL HEALTHCARE EXPENDITURES OF FREQUENT ATTENDERS: A RECORD LINKAGE STUDY BETWEEN PATIENT DATA AND REIMBURSEMENTS DATA Frans T. Smits, Henk J. Brouwer, Aeilko H. Zwinderman, Jacob Mohrs, Hugo M. Smeets, Judith E. Bosmans, Aart H. Schene, Henk C. van Weert, Gerben ter Riet BMC Fam. Pract. 2013, 14:138 ABSTRACT Background Frequently attending patients to primary care (FA) are likely to cost more in primary care than their non-frequently attending counterparts. But how much is spent on specialist care of FAs? We describe the healthcare expenditures of frequently attending patients during 1, 2 or 3 years and test the hypothesis that additional costs can be explained by FAs’ combined morbidity and primary care physicians’ characteristics. Method Record linkage study. Pseudonymised clinical data from the medical records of 16 531 patients from 39 general practices were linked to healthcare insurer’s reimbursements data. Main outcome measures were all reimbursed primary and specialist healthcare costs between 2007 and 2009. Multilevel linear regression analysis was used to quantify the effects of the different durations of frequent attendance on three-year total healthcare expenditures in primary and specialist care, while adjusting for age, sex, morbidities and for primary care physicians characteristics. Primary care physicians’ characteristics were collected through administrative data and a questionnaire. Results Unadjusted mean 3-year expenditures were 5044 and 15 824 Euros for non-FAs and three-year-FAs, respectively. After adjustment for all other included confounders, costs both in primary and specialist care remained substantially higher and increased with longer duration of frequent attendance. As compared to non-FAs, adjusted mean expenditures were 1723 and 5293 Euros higher for one-year and three-year FAs, respectively. Conclusion FAs of primary care give rise to substantial costs not only in primary, but also in specialist care that cannot be explained by their multimorbidity. Primary care physicians’ working styles appear not to explain these excess costs. The mechanisms behind this excess expenditure remain to be elucidated. 78 WHY DO THEY KEEP COMING BACK? Background Primary care physicians (PCP) spend a disproportionate amount of their time on a relatively small proportion of patients who frequently attend their practice1,2. In most studies these frequent attenders (FAs) are defined as the upper 10% of the most frequently consulting patients per sex and age group3-5. Of all FAs during one year, 15.4% continues to be a frequent attender during 3 years (1.6% of all enlisted patients)2,6. All FAs during one year (10% of all enlisted patients by definition) were responsible for 39% of all consultations while the 1.6% FAs during 3 consecutive years (persistent FAs; pFAs) were responsible for 8% of all consultations 2. FAs, and pFAs in particular, usually have multiple (chronic) somatic diseases, psychiatric disorders and social problems 2,7,8 . FAs are more frequently referred to specialist care than non-frequent attenders (non-FAs)9. However, little is known about the magnitude of the differences in healthcare utilisation and costs between non-FAs and FAs and between subgroups of FAs in specialist care. Possibly these differences in healthcare costs can be explained by specific characteristics and morbidities of FAs, and by PCP characteristics. If not, detection and treatment of underlying, not yet detected, conditions in FAs may result in a better quality of life of FAs and decrease in costs. The aim of this paper is to describe primary and specialist care costs of FAs in primary care using a combination of clinical and healthcare insurer’s data and to examine associations between healthcare expenditures of FAs of different duration in primary care and patient’s morbidities and PCP characteristics. Methods Design and data collection In a historic three-year cohort study seven primary healthcare centres in Amsterdam, The Netherlands, provided data. These centres participate in the PCP-based continuous morbidity registration network of the Department of General Practice at the Academic Medical Centre of the University of Amsterdam (HAG-net-AMC). Of all patients, 45% were insured by one health insurer: AGIS10. Only these data were used. Reimbursement claims of all insured people are electronically verified and saved at the patient level in the AGIS Health Database. This registration provides data of treatments by PCPs, specialists, other health professionals and prescriptions. Linking of both databases The two databases were linked using a number that uniquely identifies single Dutch citizens, the so-called “burger [citizen] service nummer [number]” or BSN in Dutch. Through a certified trusted third party that specializes in record linkage through irreversible pseudonymisation (ZorgTTP, Houten, The Netherlands), the PCP database (clinical data) and the insurer database (financial reimbursement data) were encrypted. Next, both encrypted databases were linked. This CHAPTER 6 79 resulted in a database in which individual patients could not be identified. Study population All patients of 18 years and older registered at the participating PCPs in 2009 were eligible for this study. Patients were classified according to their frequent attendance status. FAs were defined as those patients whose attendance rate ranked in the top 10th centile of four age groups (18–30 years; 31–45 years; 46–60 years; 61 years+) for men and women separately3. Frequent attendance was determined for each of the years 2007, 2008 and 2009. FAs during one year (1yFAs) were classified as patients who attended frequently during one of those years, FAs during 2 years (2yFAs) as patients who attended frequently in two of these years and FAs during three years (3yFAs; pFAs) as those who attended frequently during all three years. Patients who were not a frequent attender in any of these years (non frequent attenders; nonFAs) were used as a reference group. Ethics approval The study was conducted according to the Dutch legislation on data protection (Ministry of Justice, the Netherlands). Ethics approval was waived by the Medical Ethics Committee of the Academic Medical Center of the University of Amsterdam. Variables Costs In the Netherlands, health insurance covers a broad range of healthcare costs including PCP care, prescription 80 medication, specialist care in and outside hospitals and emergency care. Only over the counter medication such as simple painkillers and antihistamines are excluded. EU citizens who work and live in the Netherlands usually have Dutch healthcare insurance and were included in our study if they were AGIS insured. All Dutch citizens are required by law to have a healthcare insurance. Total costs of all reimbursed primary and specialist care costs in the years 2007–2009 were used as the dependent variable. Costs were retrieved from the insurer’s database and covered all care reimbursed to their clients during these years. By taking the sum of all three years we tried to account for fluctuating costs because of temporary diseases. We divided primary care costs in somatic costs (e.g. PCP) and psychological costs (psychologists). Costs of specialist care consist of all specialist remuneration (in and outside hospitals) and costs of hospital admissions. Mental health prescriptions have an Anatomical Therapeutic Chemical (ATC) Classification System code and are included as mental health costs, both in primary and specialist care. Costs of homecare, district nurses and nursing homes were not included. Attendance Patients were classified based on the number of face-to-face consultations with the PCP. Because we wanted to study consultation behaviour consultations with other practice staff were excluded because these contacts are almost always initiated by the PCP or his staff and concern planned control of chronic diseases. WHY DO THEY KEEP COMING BACK? Morbidity The presence of morbidity was assessed using PCPs’ registration of medical problems. A medical problem is defined as: any medical problem which needs long-term medical attention or monitoring lasting or likely to last for more than 6 months was added to the PCPs’ electronic medical record and coded according to the ICPC system11,12. These EMR data were extracted for the purpose of this study. The validity and reliability of coding of the problem list in our PCP network has been shown to be good in previous studies4;5. The problem lists were extracted at the end of 2009. We selected a set of conditions that according to the literature are associated with frequent attendance and may also be associated with costs (confounding): diabetes mellitus, cardiovascular disease, respiratory disease, cancers, locomotor problems, skin problems and digestive problems (feelings of) anxiety, (feelings of) depression, addictions, other psychological/psychiatric problems, all social problems and medically unexplained symptoms (MUS)2,4,13,14. MUS were defined according to Robbins et al. and covered several locomotor problems 15. See appendix 1. PCPs’ characteristics and practice style PCPs’ characteristics and practice style were measured using a questionnaire (Appendix 3), the mean number of registered medical problems and consultations per listed patient (adjusted for age and sex differences between the practices) and the PCP practice size (4 categories: <1000 patients; 1001–1250 patients; 1251–1500 patients; >1500 patients). Ethnicity Ethnicity of the most prevalent groups in Amsterdam (Dutch, Surinamese, Ghanese, Morrocan and Turkish) was determined in the insurer’s database using automated name recognition algorithms manually checked by employees of ethnic descent. Statistical analysis We compared primary and specialist care costs between 1yFAs, 2yFAs, pFAs and non-FAs. Associations between costs and patient characteristics were estimated using multilevel linear regression analysis with PCP as random intercept. Differences between patient groups for categorical variables were analysed using generalized linear mixed models. Statistical significance was set at P < 0.05. SPSS 20.0 for windows was used for the statistical analysis. A linear model for the actual costs implied that we modelled the mean costs. This facilitated the easy interpretation of the regression parameters, namely the cost difference associated with one unit change of the characteristic. Mean costs are also relevant for policy-makers and health insurance management because of the close relationship between average and total costs. Because the cost-distribution is highly skewed, we provided the median costs as the statistic that is better suited for individual patients. We also considered several transformations to normalize the costs distributions using the Box-Cox family of transformations, and found that a power close to zero yielded the best CHAPTER 6 81 transformation (that is, the logarithmic transformation) both for the primary and the specialist care costs16-18. The distributions of the transformed costs were however not much better than those of the untransformed costs. The results of the regression analyses were qualitatively similar for transformed and untransformed costs. Multivariate analysis was applied to determine independent predictors of costs. To determine whether PCP characteristics were associated with costs, we extended the mixed-effects models with PCP characteristics. Results Linkage Of the eligible PCP patients 2% could not be linked to the insurers’ database by missing numbers in the files of the PCPs or administrative failures. Frequent attenders In 2009 data were available on 16 531 patients. Of these patients 1208 were not enlisted in 2008 and/or 2007. Of all 16 531 patients in 2009, 2540 were classified as 1yFAs, 843 as 2yFAs, and 334 as 3yFAs, and 12 814 were non-FAs. Characteristics and medical complaints are summarized in Table 1. FAs were older and more often female than non-FAs. A non-Dutch ethnic background was slightly more prevalent among FAs. The number of all medical complaints, both somatic and psychological, was higher among FAs (in particular among pFAs) than among nonFAs. 82 Healthcare costs Median and mean costs of both primary and specialist care were significantly and substantially higher in all FA groups than in non-FAs (p < 0.001). Summed over the three years the mean primary care costs of 1yFAs, 2yFAs, 3yFAs and non-FAs were 2650, 3872, 4674 and 1645 Euros respectively. Specialist care costs showed a similar pattern: 5866, 6911, 11 150 and 3399 Euros, respectively (all differences p < 0.001). Costs that could be attributed to psychosocial complaints were much lower than costs attributed to somatic complaints in all groups but showed a similar trend over the four patient groups (p < 0.001). See Table 2. Patient-related determinants of healthcare costs Univariate associations between patient characteristics and healthcare costs and multivariate adjustment are summarized in Appendix 4 and Appendix 5. Differences between PCPs were negligible. The intraclass correlation (PCP-variance as part of the total variance) was smaller than 0.001 for both primary and specialist care. After multivariate adjustment for all patient and PCP characteristics large and significant cost differences remained between the different FA categories not only in primary care, but even more in specialist care with extra expenditures for pFAs of 1264 and 3934 Euros respectively. PCP-related determinants of healthcare costs Thirty-nine PCPs of seven primary healthcare centres participated in this study. The average practice size was 1312 WHY DO THEY KEEP COMING BACK? Table 1. Description of the study population in 2009 (by Frequent Attender-status) Non-FA1 1yFA1 2yFA1 3yFA1 Number of patients 12 814 2540 843 334 Mean age2 46 (18) 47 (18) 49 (19) 53 (17) Females, n(%) 7371 (58) 1410(56) 508 (60) 215 (64) Dutch, n(%) 9798 (77) 1872 (74) 603 (72) 235 (70) Moroccan, n(%) 465 (4) 120 (5) 49 (6) 17 (6) Turkish, n(%) 280 (2) 66 (3) 24 (3) 12 (4) Surinamese, n(%) 2262 (18) 480 (19) 167 (20) 70 (21) 3.39 (2.82) 4.50 (3.34) Ethnicity Mean number of entries on the problem list in 2009 (SD)3 All problems 1.58 (2.02) 2.43 (2.54) Social 0.02 (0.16) 0.04 (0.20) 0.06 (0.26) 0.07 (0.26) Psychological 0.14 (0.40) 0.24 (0.50) 0.35 (0.61) 0.56 (0.76) Depression 0.03 (0.18) 0.06 (0.23) 0.10 (0.30) 0.13 (0.34) Anxiety 0.02 (0.14) 0.03 (0.18) Medically unexplained symptoms 0.09 (0.33) 0.17 (0.45) 0.26 (0.54) 0.36 (0.66) Diabetes mellitus 0.09 (0.29) 0.14 (0.35) 0.18 (0.39) 0.19 (0.40) Respiratory diseases 0.13 (0.36) 0.16 (0.40) 0.25 (.47) 0.30 (0.51) Cardiovascular diseases 0.27 (0.63) 0.42 (0.74) 0.59 (.92) 0.66 (0.88) 1. 2. 3. 0.05 (0.23) 0.10 (0.32) Different frequent attender groups: Patients who never frequently attended during 3 years (non-FAs); patients who attended frequently during 1 year (1yFA), 2 years (2yFA), or 3 years (3yFA) Numbers in brackets are standard deviations, unless indicated otherwise Mean number of entries per patient on the problem list. Patient could have more than one (social, cardiovascular, etc.) problem. SD means Standard Deviation CHAPTER 6 83 patients (range, 312–2714) and 82% of PCPs participated in medical education or research. After correction for patient characteristics, intraclass correlations between healthcare costs of individual patients in the same general practice were small: 0.011 for primary care costs, 0.006 for secondary care costs. See table 4. Discussion Summary In this population of 16,531 Dutch primary care patients costs for FAs, and in particular pFAs, were considerably higher than for non-FAs throughout the healthcare. After multivariable correction for thirteen demographic and medical confounding factors at the patient and physician level, frequent attendance remained associated with higher expenditures both in primary and specialist care. Strength of this study As far as we know this study is the first to combine clinical primary care data and PCP characteristics with cost data in both primary and specialist care in this particular type of patients. Second, as most PCPs participated in the registration network for over 15 years and received regular feedback on their registration, we think these data are of good quality, especially for somatic and psychiatric (DSM-IV axis 1) problems 11. However, registration of e.g. personality disorders is expected to be less complete. Third, the cost data were collected from an insurance 84 company and are a valid reflection of the healthcare costs of the selected patients [10]. The population covered by this healthcare insurer represents the urbanized area very well10. Fourth, we used a proven method of encrypting of both databases performed by an independent third party. Of all patients only 2% could not be linked. Some of these patients may have been illegal and not insured. The distributions of patient characteristics of key interest and of the confounders guaranteed ample analytical contrast 19. For example, age varied between 18 and 101, 58% were female, and 20 percent were of Surinam origin. This resulted in enough power to robustly estimate the effect of these factors on costs. Limitations of this study Registration and coding of medical problems in primary care has limitations. In general, within a General Practice Research Network, one can distinguish several factors to explain morbidity and prescription differences: “healthcare system”, “methodological characteristics of the network”, “general practitioner”, and the “patient”. These factors and sub-factors are often interrelated and explain the different prevalence figures20,21. Because PCPs register medical problems mostly during consultations, the number of registered problems could be underreported in non-FAs (few contacts) or overreported (if resolved problems are not removed) in FAs (many contacts). Overreporting may also occur in recurrent or temporary diseases and may lead to overestimation of prevalence WHY DO THEY KEEP COMING BACK? Table 2. Median and mean 3-year costs in Euro per patient in primary and specialist care (by frequent attender status) # FA status: non 1 year 2 years 3 years Characteristics Median Mean (SD) Median Mean (SD) Median Mean (SD) Median Mean (SD) Primary care physician (PCP) 283 338 (197) 444 525 (300) 613 722 (402) 810 1005 (664) Emergency care by the PCP - 20 (164) 0 51 (114) 0 83 (156) 63 156 (334) Physical therapy1 0 41 (379) 0 77 (558) 0 99 (624) 0 181 (931) Complementary medicine1 0 16 (98) 0 24 (122) 0 37 (142) 0 43 (160) Laboratory costs 0 19 (49) 0 42 (69) 40 65 (89) 52 86 (101) psychotropic medication 0 86 (719) 0 134 (760) 0 156 (670) 16 233 (672) Antibiotics 0 15 (182) 11 28 (72) 19 40 (79) 33 63 (108) Painkillers 0 19 (165) 0 52 (358) 0 50 (305) 16 99 (296) Other medication 135 1091 (4019) 401 1717 (4724) 756 2621 (6745) 1262 2808 (4542) Somatic primary care costs 511 1553 (4212) 1049 2501 (5052) 1696 3696 (7035) 2626 4421 (5316) Psychological primary care costs 0 92 (721) 0 48 (764) 0 176 (677) 20 253 (679) All primary care costs 540 1645 (4284) 1137 2650 (5145) 1839 3872 2746 (7056) 4674 (5583) All specialist care costs 379 3399 1753 (14124) 5866 3023 (16752) 6911 5393 (12877) 11 150 (18 582) Medication: # 1 All differences between means (and medians) were statistically significant at the <0.001 level. Only the costs reimbursed by the insurer. CHAPTER 6 85 Table 3. The effects of Frequent Attender status on mean costs in primary and specialist care adjusted for all patient characteristics and morbidities (in Euros) additional costs# Primary care Specialist care Non-Frequently Attending Patients (reference)a 0 0 1-year Frequent Attenders 481 (44) 1242 (117) 2-year Frequent Attenders 800 (73) 1897 (192) 3-year Frequent Attenders 1268 (115) 4025 (302) # All effects were statistically significant at the <0.001 level. Adjusted for sex, ethnicity, age, number of active problems, diabetes, respiratory, cardiovascular, social, psychological and medically unexplained problems, cancer, locomotor, skin and digestive problems. Mean costs for non-FAs:1645 Euros (all primary care) and 3399 Euros (all specialist care). a and underestimation of the effect on costs. As in our registry, the problem lists are subject to regular evaluation, we think that the errors caused by this are likely to be small. Second, the variation in PCPs’ characteristics was modest and this reduced the data-analytic contrast at the PCP level. For example, eighty-two percent of the PCPs were involved in education or research and all practices were relatively small (mean 1,312 patients; average Dutch practice: 2,150 patients). In addition, PCPs might have tended to answer in a socially desirable way to the questions on their special interests. This may have led to a seemingly homogeneous group of PCPs and to underestimation of the effects of special interests on costs. Third, there may be undocumented determinants of costs (residual confounding). By incorporating an extensive set of possible confounders in the analysis we tried to diminish this bias. However, we had no data on severity of diseases, perceived health status, quality of life, illness attitude, life events 86 and socio-economic level. The resulting residual confounding may have led to overestimation of the association of (p) FA-ship and costs. Fourth, because 7% of the patients selected in 2009 were not enlisted all 3 years we may have slightly underestimated the number and the effect of pFAs. Fifth, because insurers compete on the basis of their premium, the patient’s choice of the AGIS health insurer could cause a some degree of selfselection. Finally, this study originates in the specific Dutch healthcare system with a well-organized primary care in which PCPs provide continuity of care and act as gatekeepers to specialist care. This may restrict the generalisability of our results to countries with a similar healthcare system like the United Kingdom[8]. Comparison with existing literature As most researchers we chose a proportional definition of frequent attendance that takes into account age and sex3-5. Earlier research has shown that the WHY DO THEY KEEP COMING BACK? Table 4. Effect of Primary Care Physicians’ characteristics on 3-year mean costs (in Euros) of primary and specialist healthcare Primary care Specialist care difference in costs (SE) difference in costs (SE) Male sex of PCP ¶, n(%) 15 (38) a -10 (20) a -100 (66) Involvement in education of medical students and/or vocational PCP training, n(%) 32 (82) -5 (36) 59 (91) 2.4 (1.1) -23 (9) -35 (32) 17 (9) -1 (1.1) -0.2 (4) Mean number of active problems per patient on problem list 1.7 (0.5) -73 (17) -289 (51) Mean number of contacts (adjusted for age and sex of the patient) 2.8 (0.3) 13 (39) -80 (131) Mean percentage of all problems that was psychological or social 12 (3) -1 (93) 2 (11) 3.2 (0.7) 0 (16) 24 (52) COPD /Asthma 3.1 (0.7) -23 (14) -50 (45) Cardiovascular disease 3.0 (0.5) -20 (18) -146 (58) Anxiety 2.9 (0.8) -11 (13) -80 (41) Depression 3.0 (0.7) -15 (14) -68 (46) Medically Unexplained Symptoms 4.0 (0.7) 10 (14) 33 (47) a General characteristicsb Practice sizec Experience (years) d Special interest in managinge Diabetes mellitus f ¶ a b c d e f PCP indicates primary care physician Numbers in brackets are standard errors, unless indicated otherwise In Euros per unit of the scale of the characteristic Divided in 4 classes: class 1:0–1000 patients; class 2:1001–1250 patients; class 3:1251–1500 patients; class 4:>1500 patients. Range 312–2,714 patients. Per (additional) year experience Five levels of interest (from 1 (no special interest) to 5 (very much interest)) and 5 levels of percentage of patients treated by the GP (0%-100%). See appendix 3. Chronic Obstructive Pulmonary Disease CHAPTER 6 87 number of health problems is consistently and positively associated with utilisation of primary and specialist care8,22. Our study confirms these findings and evaluates the influence of clinical and physician characteristics on the relationship between frequent attender status and healthcare costs. The mechanism behind the interaction between FAs and PCP prompting the performance of additional diagnostic and therapeutic actions is not fully understood. Earlier theories emphasized a negative interaction between some patients and their PCP in maintaining “somatic fixation” resulting in unnecessary consultations, tests and treatments 2325 . In patients with MUS explanations for ‘somatisation’ should be sought in doctor-patient interaction rather than in patients’ psychopathology26-28. However, the adjusted differences in costs between the PCPs participating in this study were small. This suggests that patient determinants may be more important in explaining extra expenditures by FAs than PCP characteristics, although a larger and more diverse group of PCPs may be needed to corroborate this. The nestor of Dutch general practice, Frans Huygen, already demonstrated that families tend to be consistent in illness and consultations patterns29-31. This may imply that frequent attending also could be understood as a kind of learned behaviour or trait. Implications for research and/or practice This study shows that FAs of the PCP are also heavy users of all clinical services. As most somatic problems in 88 this patient group are already dealt with in chronic care models, most advantages are likely to be gained by diagnosing and treating undetected psychiatric and social problems. Adequate diagnosis and treatment of such problems in primary care and optimal PCP-patient-communication may prevent referral of patients to specialist care and may strengthen the gate keeping role of PCPs32-36. The assumed trait character of frequent attendance may impede the effects of any treatment. Future research should focus on the aetiology of (the persistence of) frequent attendance 36,37. Personality factors, life events, social support and socio-economic level should be investigated more to assess how they affect attendance and costs. Moreover, we need to clarify how and why patients prompt their physicians to do more than strictly indicated. Armed with more knowledge about these causes of frequent attendance future randomised trials may target those interventions aimed at modifying these causes and reduce illness, attendance and costs. Conclusions Frequent attenders of primary care contribute substantially to costs not only in primary care but also in specialist care. Morbidity, social problems and PCP characteristics appear to only partly explain these expenditures. Frequent attendance may therefore be considered as an independent, yet incompletely understood contributing determinant of healthcare utilisation and costs in primary and specialist care. WHY DO THEY KEEP COMING BACK? References 1. Neal RD, Heywood PL, Morley S, Clayden AD, Dowell AC: Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br J Gen Pract 1998, 48:895– 898. 2. Smits FT, Brouwer HJ, Ter Riet G, Van Weert HC: Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC Publ Health 2009, 9:36. 3. Smits FT, Mohrs J, Beem E, Bindels PJ, Van Weert HC: Defining frequent attendance in general practice. BMC Fam Pract 2008, 9:21. 4. Vedsted P, Christensen MB: Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005, 119:118–137. 5. Luciano JV, Fernandez A, Pinto-Meza A, Lujan L, Bellon JA, Garcia-Campayo J, et al: Frequent attendance in primary care: comparison and implications of different definitions. Br J Gen Pract 2010, 60:49–55. 6. Smits FTM, Brouwer HJ, Ter Riet G, Van Weert HC: Predictability of persistent frequent attendance. A historic 3-year cohort study. Br J Gen Pract 2009 2009, 2:114–119. 7. Vedsted P, Fink P, Sorensen HT, Olesen F: Physical, mental and social factors associated with frequent attendance in Danish general practice. A populationbased cross-sectional study. Soc Sci Med 2004, 59:813–823. 8. Dunlop S, Coyte PC, McIsaac W: Socioeconomic status and the utilisation of physicians’ services: results from the Canadian National Population Health Survey. Soc Sci Med 2000, 51:123–133. 9. Heywood PL, Blackie GC, Cameron IH, Dowell AC: An assessment of the attributes of frequent attenders to general practice. Fam Pract 1998, 15:198–204. 10. Smeets HM, De Wit NJ, Hoes AW: Routine health insurance data for scientific research: potential and limitations of the Agis Health Database. J Clin Epidemiol 2011, 64:424–430. 11. Brouwer HJ, Bindels PJ, Van Weert HC: Data quality improvement in general practice. Fam Pract 2006, 23:529–536. 12. Lamberts H: Wood M e: International classification of primary care. Oxford: Oxford University Press; 1988. 13. Gill D, Sharpe M: Frequent consulters in general practice: a systematic review of studies of prevalence, associations and outcome. J Psychosom Res 1999, 47:115–130. 14. Ferrari S, Galeazzi GM, Mackinnon A, Rigatelli M: Frequent attenders in primary care: impact of medical, psychiatric and psychosomatic diagnoses. Psychother Psychosom 2008, 77:306–314. 15. Robbins JM, Kirmayer LJ, Hemami S: Latent variable models of functional somatic distress. J Nerv Ment Dis 1997, 185:606–615. 16. Box GEP, Cox DR: An Analysis of Transformations. Journal of the Royal Statistical Societ 1964, 26(2):211–256. 17. Manning WG, Mullahy J, Basu A: Estimating log models: to transform or not to transform? J Health Econ 2001, 20:461–494. 18. Manning WG, Basu A, Mullahy J: Generalized modeling approaches to risk adjustment of skewed outcomes data. J Health Econ 2005, 24:465–488. 19. Miettinen OS: Theoretical Epidemiology: principles of occurrence research in medicine. New York: Wiley; 1985. 20. Van den Dungen C, Hoeymans N, Gijsen R, van den Akker M, Boesten J, Brouwer H: What factors explain the differences in morbidity estimations among general practice registration networks in the Netherlands? A first analysis. Eur J Gen Pract. 2008;14 Suppl 1:53-62. CHAPTER 6 89 21. van den Dungen C, Hoeymans N, Boshuizen HC, van den Akker M, Biermans MC, Van BK: The influence of population characteristics on variation in general practice based morbidity estimations. BMC Publ Health 2011, 201(1):887. 22. Wouterse B, Meijboom BR, Polder JJ: The relationship between baseline health and longitudinal costs of hospital use. Health Econ 2011, 20(8):985–1008. 23. Van Eijk J, Grol R, Huygen F, Mesker P, Mesker-Niesten J: [Somatic fixation. Prevention by the general practitioner]. Soc Sci Med 1982,58:717–725. 24. Rosendal M, Fink P, Bro F, Olesen F, Rosendal M, Fink P, et al: Somatization, heartsink patients, or functional somatic symptoms? Towards a clinical useful classification in primary health care. Scand J Prim Health Care 2005, 2005:3– 10. 25. McDaniel SH, Campbell T, Seaburn D: Treating Somatic Fixation: A Biopsychosocial Approach: When patients express emotions with physical symptoms. Can Fam Physician 1991, 37: 451-456. 26. Salmon P, Humphris GM, Ring A, Davies JC, Dowrick CF, Salmon P: Primary care consultations about medically unexplained symptoms: how do patients indicate what they want? Psychosom Med 2006, 24:570–577. 27. Ring A, Dowrick CF, Humphris GM, Davies J, Salmon P, Ring A: Doctors’ responses to patients with medically unexplained symptoms who seek emotional support: criticism or confrontation? Soc Sci Med 2005, 29:1505–1515. 28. Ring A, Dowrick CF, Humphris G, Salmon P: What do general practice patients want when they present their medically unexplained symptoms, and why do their general practitioners feel pressurized? J Psychosom Res 2005, 59:255–260. 30. van den Bosch WJ, Huygen FJ, van den Hoogen HJ, Van Weel C: Morbidity in early childhood: family patterns in relation to sex, birth order, and social class. Fam Med 1993, 1993:126–130. 31. Cardol M, van den Bosch WJHM, Spreeuwenberg P, Groenewegen PP, Van Dijk L, De Bakker DH: All in the Family: Headaches and Abdominal Pain as Indicators for Consultation Patterns in Families. The Annals of Family Medicine 2006, 4:506–511. 32. Cuijpers P, Van Straten A, Schuurmans J, Van Oppen P, Hollon S, Cuijpers P: Psychotherapy for chronic major depression and dysthymia: A metaanalysis. Clin Psychol Rev 2010, 30: 51-62. 33. Cuijpers P, Van Straten A, Warmerdam L: Problem solving therapies for depression: A meta-analysis. European Psychiatry 2007, 22:9–15. 34. Reger MA, Gahm GA: A meta-analysis of the effects of Internet- and computerbased cognitive-behavioral treatments for anxiety. J Clin Psychol 2009, 65(1):53–75. 35. van der Feltz-Cornelis CM, Van Os TW, Van Marwijk HW, Leentjens AF.: Effect of psychiatric consultation models in primary care. A systematic review and meta-analysis of randomized clinical trials. J Psychosom Res 2010, 68:521-33. 36. Morriss R, Kai J, Atha C, Avery A, Bayes S, Franklin M: Persistent frequent attenders in primary care: costs, reasons for attendance, organisation of care and potential for cognitive behavioural therapeutic intervention. BMC Fam Pract 2012, 13:39. 37. Rifel J, Svab I, Selic P, Rotar PD, Nazareth I, Car J: Association of Common Mental Disorders and Quality of Life with the Frequency of Attendance in Slovenian Family Medicine Practices: Longitudinal Study. PLoS One 2013, 8:e54241. 29. Huygen FJ: Family Medicine- The medical life history of families. Assen, The Netherlands: van Gorcum; 1978. 90 WHY DO THEY KEEP COMING BACK? CHAPTER 6 91 part II REVIEW OF THE LITERATURE ABOUT INTERVENTIONS ON FREQUENT ATTENDERS IN PRIMARY CARE chapter 7 INTERVENTIONS ON FREQUENT ATTENDERS IN PRIMARY CARE. A SYSTEMATIC LITERATURE REVIEW Frans T. Smits, Karin A. Wittkampf, Aart H. Schene, Patrick J. Bindels, Henk C. van Weert Scand. J. Prim. Health Care. 2008; 26(2): 111-6. ABSTRACT Purpose To analyze which interventions are effective in influencing morbidity, quality of life and healthcare utilization of frequently attending patients (FAs) in primary care. Method We performed a systematic literature search for articles describing interventions on FAs in primary care (Medline, Embase and PsycINFO). Outcomes were morbidity, quality of life and use of health care. Two independent assessors selected all randomized clinical trials (RCT) and assessed the quality of the selected RCTs. Results Three RCTs used frequent attendance to select patients at risk for distress, major depression and anxiety disorders. These RCTs applied psychological and psychiatric interventions and focused on as yet undiagnosed psychiatric morbidity of FAs. Two of them found more depression-free days and a better quality of life after treating Major Depressive Disorder (MDD) in FAs. No other RCT found any positive effect on morbidity or quality of life. Two RCTs studied an intervention which focused on reducing frequent attendance. No intervention significantly lowered attendance. Due to the difference in study settings and the variation in methods of selecting patients, meta-analysis of the results was not possible. Conclusion We did find indications that frequent attendance might be a sign of as yet undiagnosed MDD and that treatment of MDD might improve the depressive symptoms and the quality of life of depressed FAs. We found no evidence that it is possible to influence healthcare utilization. Future studies should focus on well-defined subgroups of FAs. 94 WHY DO THEY KEEP COMING BACK? Introduction Primary care physicians (PCP) spend about 80% of their time on 20% of their patients: about one in every seven consultations concerns the top 3% of attenders.1 Two systematic literature reviews confirm that these frequent attenders (FAs) have high rates of physical disease, emotional distress, psychiatric illness and social difficulties.2;3 Frequent attenders are a heterogeneous group of patients. Karlsson’s analysis of FAs suggests dividing them into five subgroups: patients with purely somatic illness (28%), patients with clear psychiatric illness (21%), patients in temporary crisis (10%), chronically somatizing patients (21%) and those with multiple problems (20%).4 In most cases somatic and psychiatric illnesses are accepted reasons for frequent consultation, crises pass and are a reason for frequent consultation for a short time. However frequent attendance by multi-problem or somatizing patients with related but undetected psychiatric morbidity is thought to lead to unnecessary consultations and therefore to ineffective health care.5 Detecting, diagnosing and treating the psychiatric disorders of these FAs should improve their quality of life as well as lower the impact of frequent attending on the healthcare system.6 The combination of large workload and high rate of (chronic) disease make FAs an important group for a PCP not only to study but also to treat. The objective of this study is to analyze which interventions might be helpful in reducing morbidity and improving the quality of life of FAs and might reduce their healthcare utilization. We therefore performed a systematic review of interventions on FAs aimed at answering two questions: (1) which interventions have been studied, and at which group of FAs were they targeted, and (2) what was the effectiveness of these interventions in terms of morbidity, quality of life and frequency of attendance? Methods Literature search: We searched the databases Medline, Embase and PsycINFO (1980- 2006-11). To obtain optimal sensitivity we used the MESH-headings: ‘health services/ utilization’, ‘health services misuse’ and ‘health care utilization’, as well as the following truncations as text words: ‘frequent attend*’, ‘frequent consult*’, ‘high utiliz*’, ‘high consultation frequency’,’ high consultation rate’. In addition we checked the references of all included articles for other relevant but not yet retrieved articles. Selection of articles: On title and abstract, we selected articles which described interventions in FAs in primary care aged between 18 and 70 years, and were written in the English, French, Dutch or German language. We included all possible FA-definitions, also definitions based on specific (sub) groups of primary care patients. When there was any doubt about the setting or the kind of included patients we assessed the full paper. An overview of the FA definitions used in the final selection of articles for this review is presented in table 1. CHAPTER 7 95 All articles that met the inclusion criteria were read in detail to select only randomized controlled trials (RCT) and to re-check the inclusion and exclusion criteria. Two assessors (FS; KW) performed these procedures independently and the final selection was discussed in a consensus meeting with a third assessor (HW). Quality assessment: Two reviewers (FS and KW) appraised each RCT independently with the quality criteria for assessment of experimental studies of Khalid Khan et al.7 This checklist consists of nine items on methodological quality. All items were scored as yes, no or uncertain. Points of disagreement were discussed with a third senior assessor (HW) for a final decision. Because of the differing study settings and the variation in studied populations, pooling of the results was not possible. Results Literature search: Our literature search resulted in 4357 articles. After the first selection, 28 articles were retrieved for detailed reading. (Figure 1) The second selection resulted in the identification of five RCTs. Table 1 presents an overview of their characteristics. Included studies: Setting, definition of FAs, kind of counted contacts and the population of the included studies are summarized in table 1. Simon et al. and Katzelnick et al. refer to the same research program. 8; 9 Their main goal was the evaluation of a depression management program among depressed 96 FAs. They excluded patients who had received active treatment for depression during the previous three months and for whom the treatment program would be inappropriate (i.e. bipolar or psychotic disorder, substance abuse or terminal illness). The selected patients (n=7203) were screened by telephone using the depression module of the Structured Clinical Interview for DSM-IV (SCID). Those currently suffering from major depression or those reporting an episode of major depression within the previous two years but now in partial remission (n=1475) were eligible for a second assessment using the Hamilton Depression Rating Scale (HDRS). Finally, a total of 407 patients with a HDRS score of 15 or more consented to enrolment: 218 patients were randomized to a Depression Management Program (DMP) and 189 patients received usual care (UC). Intervention: The depression management program included a two hour physician training program, an evaluation visit with their PCP immediately after enrolment, antidepressant medication (AD) if appropriate, written and videotaped educational materials and treatment coordination. Results: in the year following the intervention patients in the intervention group had a mean of 47 more depression-free days (c.i. 26.6-68.2 ), more prescriptions for AD (69.3% of DMP patients and 18.5% of UC patients had filled at least three antidepressant prescriptions for the six-month period after enrolment ; P<0.001), more improvement on the HDRS (change in HDRS in 12 months for DMP patients 9.2 and for UC patients 5.6; P< 0.001), more improvement on the SF- WHY DO THEY KEEP COMING BACK? Figure 1. Flow diagram selection of articles Articles about frequent attending: N= 4357 (Pubmed, Embase, PsycINFO) Articles about interventions on frequent attenders: N= 28 Exclusion criteria (N= 4329): • not about interventions on FAs(N= 4329) • paediatric- or geriatric-care (n= 0) Exclusion criterion (N= 23): • no RCT. Final list of RCT’s on frequent attenders: N= 5 Analyses 20-scores for social functioning, mental health, and general health perceptions (P<0.05), and DMP patients had 3.2 more contacts with the healthcare system (c.i. 0.70-5.80) as well as more costs ($51.84 per additional depression-free day ; c.i. 17.37108.47). Katon et al. evaluated a psychiatric consultation-liaison program among distressed FAs. 8 They selected 1790 FAs (235 patients were excluded for various reasons). From this group distressed FAs were selected by using the Symptoms Checklist Revised (SCL-R); sum score 1 standard deviation above population mean. Of the 339 identified distressed FAs, 251 gave consent for randomization, 124 patients were assigned to the intervention group and 127 to the control group. The intervention consisted of a psychiatric diagnostic interview by a psychiatrist using the Diagnostic Interview Schedule (DIS) with the family physician present, a jointly formulated treatment plan and a mutually accepted course of action (i.e. medication adjustment, referral, fixedinterval visits etc.). The outcome measures were rates of anxiety and depression (SCL-90-R), use of antidepressants and use of health care. Results: Katon found no significant difference in improvement of psychopathology, more prescribed antidepressants (+ 38%; p<0.01.) in the intervention group after one year and no consistently significant differences in any utilization measure between intervention and control groups (primary care, p= 0.097; medical specialty visits, p=0.111; radiography, p= 0.61; lab testing, p=0.072; admission to inpatient care, p=0.16). . CHAPTER 7 97 Table 1. Overview of the selected Randomized Clinical Trials Setting Definition FAs/ Kind of counted contacts Population Identification Number of interventions / contr. patients Simon 8* Primary care clinic, 3 prepaid health plans in Midwest, Northwest and New England, (USA) • Top 15% attenders during 2 consecutive years • outpatient medical visits Age: 23-63 • Electronic data • SCID : Pos MDD or MDD pos. last 2 yrs (=1475 pat) • HDRS>14, 163 General practices: • Usual care: 81 • Intervention: 82 Intervention: 218 Usual care: 189 • Top15% attenders during 2 consecutive years • outpatient medical visits Age: 25-63 • Electronic data • SCID : Pos MDD or MDD pos last 2 yrs (=1475 pat) • HDRS>14 ,163 General practices: • Usual care: 81 • Intervention: 82 Intervention 218 Usual care 189 • Top 10% attenders in 1 year for sex and age. • ambulatory health care visits. Age: 18-75 • >2 Years in practice • Selection from electronic data • SCL-R one standard deviation above mean -> 339 patients. • 251 Accepted randomisation Intervention 124 Control 127 Intervention 34 Control 40 No contact 30 Katzelnick 9 Primary care clinic, 3 prepaid health plans in Midwest, Northwest and New England, (USA) Katon 10 Primary care clinics of HMO, Washington State, (USA) 18 GP’s out HMO 300.000 Patients. Olbrisch 11 Primary health care for students: Florida State University, (USA) • > 4 face-to-face contacts in first quarter of study year • outpatient medical visits Freshmen, sophomores and juniors: students university health centre • • • • • 400 Students 300 got letter 129 agreed 112 randomized Plus “no contact”-group. • > 4 Out-of-hours contacts one year before inclusion. • consultations, home visits and telephone calls No age restriction. • Consecutive patients • Randomizat. per practice. Christensen12 Primary care outof-hours service, County of Northern Jutland, (Denmark) SCID MDD HDRS SF 98 Structured Clinical Interview DSM Major Depressive Disorder Hamilton Depression Rating Scale Social Functioning WHY DO THEY KEEP COMING BACK? Intervention practices: 83 GP’s; 3500 patients Control practices: 93 GP’s 4635 patients Intervention Follow up Outcomes Results • Depression management program(DMP): • 2 h training • evaluation contact • antidepressant medication • information material • treatment coordinator 1 Year after randomization. • Depression free days • Costs • More depression free days (229>182) • More costs (+$51.84 per additional depression free day) • Depression management program(DMP): • 2 h training • evaluation contact • antidepressant medication • information material • treatment coordinator 1 Year after randomization. • HDRS • SF-20 score • Use of antidepressant medication • Attendance • Improvement HDRS (13,6-> 9,9 at 1 year) • More use of AD (69.3% of DMPpatients and 18.5% of usual-carepatients with at least 3 prescriptions in 0, 5 year). • Better SF-20 scores for social funct, mental health, gen. health perceptions • More attendance in year after inclusion (+3, 2 ) • DIS by psychiatrist • Interview by the psychiatrist with the GP present • Jointly formulated treatment plan • Written protocol of treatment for GP 1 Year after randomization • Use of antidepressant med (AD). • Rate of anxiety/ depression • Use (psych) health care • More AD (+38%) • No better psych state • No lower use of health care and costs Brief educational program (group of 3-8 students) 1 Year after intervention • Number of contacts primary care • Use of other health care • Lower use of primary care on short term. • Convergence towards same utilization during follow-up • No differences on number of visits to other health care providers • Status consultation by GP • Education of participating GP’s • Questionnaire patients • Economic incentives GP 1 Year after the intervention • Number of contacts with the outof-hours-service. • Daytime contacts with the GP;hospital admissions; visits to hospital outpatients clinics • No convincing effect. SCL-R DIS * Symptom Checklist Revisited Diagnostic Interview schedule The numbers in superscript refer to the reference list CHAPTER 7 99 Table 2. Quality assessment selected RCTs Quality criteria (7*) Simon Katzelnick (8) (9) Katon Olbrisch Christensen (5) (12) (13) Assignment to the treatment groups really random? + + + ? + Treatment allocation concealed? + + ? ? - Groups similar at baseline in terms of prognostic factors? + +a + + ? Were the eligibility criteria specified? + + + + + Outcome assessors blinded to the treatment allocation? + + + - n.a. Was the care provider blinded? - - - - - Was the patient blinded? - - - - - Points estimates and measure of variability presented for the primary outcome measure? + + + - - Analyses included an intention to treat analysis? +a + - - - a * Not mentioned in this article. Katzelnick does mention the criteria. Numbers between brackets refer to the reference list Olbrisch refers to an intervention among frequent attending students. 9;10 Her purpose was to evaluate the effectiveness of a brief health education intervention aimed at making students aware of the psychological and social factors that make people prone to illness and to inappropriate use of health care resources. Three-hundred randomly-selected and eligible students were sent a letter inviting them to participate, 129 agreed and 112, who kept appointments scheduled for them, were randomized to the intervention group (n=34) or the control group (n=40). Olbrisch also selected a matched group with no contacts (n=30). The exact routing of all study participants 100 is not clearly described. Her intervention consisted of a brief educational group program (presentations, discussion and a demonstration or audiotape of deep muscle relaxation). The outcome measure was use of health care facilities. Results: the intervention group showed reduced utilization of the university health center for a short period of time (not adequately specified), with this effect dissipating over time and no significant differences on the number of visits to other health care providers (F(2.85)=1.7 ; p=0.19). In the only RCT outside of the USA, Christensen et al studied an out-of-hours primary care service in Denmark.11 WHY DO THEY KEEP COMING BACK? They tested whether a combination of intervention strategies reduced health care utilization by FA’s.. In a cluster randomization, family physician practices were randomized to intervention practices (83 practices; 3500 patients) and control practices (93 practices; 4635 patients). The intervention consisted of (1)a patient questionnaire and an invitation for the FAs to contact their family physician for a status consultation, (2)information about the project and FAs for the PCP,(3) physician group education on frequent attending (29% of all physicians representing 40% of all practices participated) and (4)economic incentives for the PCP to perform the status consultation. Outcome measures were (1) the number and kind of contacts with the out-of-hours-service (2) daytime contacts with PCP, hospital admissions, and visits to hospital outpatient clinics and emergency departments. Results: They found no significant difference in the primary and secondary outcome measures. Quality assessment The quality assessment of the included RCTs is summarized in Table 2. None of the RCTs fully complied with all quality criteria. In none of the RCTs were the patient and the care provider sufficiently blinded. Blinding of patients and physicians was not possible in the studies of Simon, Katzelnick, Olbrish and Katon because psychological treatments do not allow concealment. Katon, Olbrisch and Christensen did not include an intentionto-treat-analysis. Olbrisch did not describe whether the outcome assessors were blinded to the treatment allocation and did not give point estimates and measures of variability. Christensen did not go into detail about point estimates and measures of variability. All articles, except Christensen’s, refer to various subgroups of FAs. Therefore it was not possible to generalize the results of these studies to all FAs. Discussion Main findings After an extensive search for all relevant literature we were able to identify five randomized controlled trials that studied interventions on FAs. Our aim was to learn more about the included FA population, about the type of intervention program and its effectiveness in improving morbidity and quality of life and in lowering attendance. The outcome of these interventions was disappointing. We found just 5 primary care based trials. The populations under study as well as the outcomes of studies differed. Two RCTs found more depression-free days and reported a better quality of life after treating MDD in a subgroup of depressed FAs.12;13 Although on an individual basis the gain was quite impressive (a mean of 47 depression free days) the net gain on a group level was disappointing: for every SCID 2.6 depression free day could be achieved. One other RCT found no positive effect on morbidity.8 Only two RCTs included clear measures of quality of life.12;13 Finally, all RCTs concluded that the studied interventions did not significantly lower attendance during one year of follow-up. CHAPTER 7 101 Two RCTs describing an intervention in depressed FAs found even more contacts, more prescriptions for AD and more costs in the intervention group within one year of follow-up. 12;13 One RCT found more prescription of AD and no significant difference in health utilization.8 Two RCTs did not measure costs.9;11 Strength and limitations An important limitation was the differences in study settings and the variation in methods of selecting patients. Four studies were carried out in the USA (3 HMO; 1 university healthcare), one in Denmark (out-of-hours-service). We also found that frequent attendance is not a clearly-defined concept. Two studies selected patients who were FA for two consecutive years (Simon, Katzelnick). Other studies selected patients who were FA for three months (Olbrisch) or one year (Katon, Christensen).Three studies selected a percentile of most attending patients; two used a certain number of consultations as a selecting criterion. In three studies, frequent attendance was used to select a group of patients at risk for distress, major depression and/or anxiety disorders.8;12;13 The other two made no further selection and intervened in all FAs.9;11 Also the interventions used were different: in three studies, which focused on as yet undiagnosed psychological problems and psychiatric morbidity of FAs, interventions consisted of a screening and depression management program and a treatment plan and intervention by a psychiatrist. 8;12;13 One used an educational group program.9 In only one study the intervention (mainly a status consultation 102 and incentives for the PCP) was carried out by a PCP and focused on diminishing attendance.11 Due to the low number of PCP’s trained in this study, it is likely that the success of this intervention was underestimated. Because of all these differences we cannot generalize results to other (sub) group of FAs. A possible explanation for the lack of generalisibility could be that frequent attendance is the result of many diseaseand personality-linked factors which make frequent attenders a heterogeneous group of patients.4 Intervening on a specific aspect of frequent attendance, for instance depression, dilutes the outcome of a RCT which studies all FAs. Moreover, frequent attendance is not a consistent personality trait, but often a transitory characteristic. Some studies show that up to 60-70% of frequently attending patients change their health-seeking behavior within 2-3 years. 14-17 Using healthcare utilization as an outcome measure therefore does not seem adequate in studying FAs, defined on a one year basis. Studies that did find an effect used consultation patterns on a two year basis.12;12 When an intervention is planned the net effect on healthcare utilization in the short term logically is upwards and a follow-up of longer than one year is needed. Our study is the first that reviews interventions on FAs. The strength of this study is the sensitive search with both Mesh-headings and text words. We therefore expect not to have missed any RCT describing an intervention in FAs. WHY DO THEY KEEP COMING BACK? Comparison with relevant literature There is an extensive literature about the characteristics of (sub) groups of FAs. There are little studies (n= 28) which try to influence morbidity, quality of life and use of healthcare of FAs. Only five are RCT’s. Definitions of FAs differed considerably. We propose to follow the advice of Vedsted et al.to define FAs as the top 10% of all enlisted patients.2 Conclusion We found a small number of studies that evaluated interventions on FAs. There is no evidence that it is possible to influence healthcare utilization by frequent attenders. Treatment of (not yet diagnosed) major depressive disorder might improve the symptoms and the quality of life of depressed FAs, but will not reduce their consultation rate within one year of followup. References (1) Neal RD, Heywood PL, Morley S, Clayden AD, Dowell AC. Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br J Gen Pract 1998; 48(426):895-898. (2) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (3) Gill D, Sharpe M. Frequent consulters in general practice: a systematic review of studies of prevalence, associations and outcome. J Psychosom Res 1999; 47(2):115-130. (4) Karlsson H, Joukamaa M, Lahti I, Lehtinen V, Kokki-Saarinen T. Frequent attender profiles: different clinical subgroups among frequent attender patients in primary care. J Psychosom Res 1997; 42(2):157-166. (5) Katon W, von Korff M, Lin E, Lipscomb P. Distressed high utilizers of medical care: DSM-III--R diagnoses and treatment needs. General Hospital Psychiatry /11; 12(6):355-362Record. (6) Dowrick CF, Bellon JA, Gomez MJ. GP frequent attendance in Liverpool and Granada: the impact of depressive symptoms. Br J Gen Pract 2000; 50(454):361-365. (7) Khan KS, ter Riet G, Popay J, Nixon J, Kleijnen J. Study quality assessment. In Undertaking Systematic Reviews of Research on Effectiveness CRD’s Guidance for Carrying Out or Commissioning Reviews.2 . 2001. York: NHS Centre for Reviews and Dissemination (CRD), University of York. Ref Type: Generic (8) Katon W, von Korff M, Lin E, Bush T. A randomized trial of psychiatric consultation with distressed high utilizers. General Hospital Psychiatry /3; 14(2):86-98Record. CHAPTER 7 103 (9) Olbrisch ME. Evaluation of a stress management program for high utilizers of a prepaid university health service. Dissertation Abstracts International /5; 39(11-B):5573Record-1982. (10) Olbrisch ME. Evaluation of a stress management program for high utilizers of a prepaid university health service. Med Care 1981; 19(2):153-159. (11) Christensen MB, Christensen B, Mortensen JT, Olesen F. Intervention among frequent attenders of the outof-hours service: a stratified cluster randomized controlled trial. Scand J Prim Health Care 2004; 22(3):180-186. (12) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (13) Simon GE, Manning WG, Katzelnick DJ, Pearson SD, Henk HJ, Helstad CP. Costeffectiveness of systematic depression treatment for high utilizers of general medical care. Archives of General Psychiatry /2; 58(2):181-187. (14) Ward AM, Underwood P, Fatovich B, Wood A. Stability of attendance in general practice. Fam Pract 1994; 11(4):431-437. (15) Andersson S-O, Lynoe N, Hallgren C-G, Nilsson M. Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scandinavian Journal of Primary Health Care 2004; 22(2):91-94. (16) Carney TA, Guy S, Jeffrey G. Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 2001; 51(468):567-569. (17) Botica MV, Kovacic L, Tiljak MK, Katic M, Botica I, Rapic M et al. Frequent attenders in family practice in Croatia: Retrospective study. Croatian Medical Journal 2004; 45(5):620-624. 104 WHY DO THEY KEEP COMING BACK? CHAPTER 7 105 part III A PROSPECTIVE STUDY OF FREQUENT ATTENDERS chapter 8 WHY DO THEY KEEP COMING BACK? PSYCHOSOCIAL AETIOLOGY OF PERSISTENCE OF FREQUENT ATTENDANCE IN PRIMARY CARE: A PROSPECTIVE COHORT STUDY Frans T. Smits, Henk J. Brouwer, Aeilko H . Zwinderman, Jacob Mohrs, Aart H. Schene, Henk C.P.M. van Weert, Gerben ter Riet Accepted by the Journal of Psychosomatic Research ABSTRACT Background Patients who visit their General Practitioner (GP) very frequently over extended periods of time often have multi-morbidity and are costly in primary and specialist healthcare. We investigated the impact of patient-level psychosocial and GP-level factors on the persistence of frequent attendance (FA) in primary care. Method Two-year prospective cohort study in 623 incident adult frequent attenders (>90th attendance centile; age and sex-adjusted) in 2009. Information was collected through questionnaires (patients, GPs) and GPs’ patient data. We used multilevel, ordinal logistic regression analysis, controlling for somatic illness and demographic factors with FA in 2010 and/ or 2011 as the outcome. Results Other anxiety (odds ratio (OR) 2.00; 95% confidence interval from 1.29-3.10) over 3 years and the number of life events in 3 years (OR 1.06; 1.01-1.10 per event; range of 0 to 12) and, at baseline, panic disorder (OR 5.40; 1.67-17.48), other anxiety (OR 2.78; 1.04-7.46), illness behaviour (OR 1.13; 1.05-1.20 per point; 28-point scale) and lack of mastery (OR 1.08; 1.01-1.15 per point; 28-point scale) were associated with persistence of FA. We found no evidence of synergistic effects of somatic, psychological and social problems. We found no strong evidence of effects of GP characteristics. Conclusion Panic disorder, other anxiety, negative life events, illness behaviour and poor mastery are independently associated with persistence of frequent attendance. Effective intervention at these factors, apart from their intrinsic benefits to these patients, may reduce attendance rates, and healthcare expenditures in primary and specialist care. 108 WHY DO THEY KEEP COMING BACK? Introduction Some patients have exceptionally high consultation rates with their General Practitioner (GP), sometimes over many years.1-5 These persistent frequent attenders (FAs) often have multi-morbidity and are costly in both primary and specialist healthcare.6 Anyone may have short periods in her/ his lives in which frequent help from a GP is sought or needed. However, when such periods exceed two or more consecutive years, more structural psychosocial problems are often present.1;7;8 Thus, persistent frequent attendance could be seen as an easily detectable marker for underlying, often undetected and unmet, psychosocial problems or diseases. Previous work has shown that psychological distress, low physical quality of life and low educational level were associated with persistence of frequent attendance (when frequent attendance is proportional defined).9;10 Using a fixed cutoff, persistence of frequent attendance has been found to be associated with such diverse factors as female gender, obesity, former frequent attendance, fear of death, alcohol abstinence, low satisfaction, and irritable bowel syndrome.11 Because high attendance is strongly related to age and sex, we think it makes sense to define frequent attendance as an age and sexadjusted attendance rate ranking in the top 10 centile in a GP-practice within a time frame of one year.12;13 Such a proportional threshold definition selects the exceptionally high utilizers within each age and sex group and allows meaningful comparison between practices, periods, and countries. However, the literature on the precise aetiology of persistence of frequent attendance is equivocal.7;11;14-18 Theoretically, within a specific healthcare setting attendance may be influenced by patient characteristics including morbidity, by GP characteristics like work style, experience, personality and professional interests and thirdly by the interpersonal dynamics between patients and their physician .19;8;16 Previous work indicates that the considerable costs in primary as well as in specialist care associated with (persistent) frequent attendance may not simply be explained by the excess morbidity these patients have.20 Therefore, even fully effective treatment, of clearcut multi-morbidity, if available, cannot be expected to reduce to normal the high attendance rates and associated referral rates to specialist care. The incomplete understanding of the aetiology of persistence of frequent attendance hampers the thoughtful design of preventive strategies for this persistence and risks any trials to be well-intentioned shots in the dark. Therefore we followed over 2 years a cohort of frequent attenders during one year to investigate which and to what extent psychological and social factors, and GP characteristics play a role in the aetiology of persistence of frequent attendance. Methods Design In a prospective cohort study, we followed incident FAs for two consecutive years. CHAPTER 8 109 Box. Key variables and confounding variables (continued on right page) Scale Structure Range* Illness Attitude Scale 27 questions; 5 point scale 0-108 Health anxiety 11 questions; 5 point scale 0-44 Illness behaviour 6 questions; 5 point scale 0-24 Mastery Pearlin-Schooler Mastery Scale 7 questions; 5 point scale 0-28 Life events last 12 months numeric Number of life events 0-15 # Depression Patient Health Questionnaire 9 items of the DSM IV (algorithm)1 y/n Variable Key variables Somatoform problems Severity score 2 0-27 y/n y/n Panic disorder Patient Health Questionnaire First screening question positive and >2 positive scores on the second panic question3 Other anxiety Patient Health Questionnaire First screening question positive and >4 positive scores on the second anxiety question4 Confounding variables Somatic morbidity5 Diabetes y/n Any chronic respiratory disease y/n Any chronic cardiovascular disease y/n Any cancer problem y/n Any locomotor problem y/n Any skin problem y/n Any digestive problem y/n Social problems5 Any social problem y/n Psychological problems5 Any psychological problem y/n Medically Unexplained Symptoms5 Any medically unexplained symptom (according to Robbins6) y/n Demographic patient data Age Linear Sex Male; female Education 7 levels of education 1-7 Ethnicity Dutch y/n Surinamese y/n Other y/n Living situation Living alone (with small children) y/n Employment Paid work or study y/n Body Mass Index 110 18- 93 # Weight (kg)/ height (m) WHY DO THEY KEEP COMING BACK? 2 16-48 # Structure Range* Practice size <1000; 1001-1250; 1251-1500; >1500 patients 1-4 Special knowledge of/interest in a specific part of general practice Yes or no y/n Variable Scale GP characteristics Paediatric medicine y/n Geriatric medicine y/n Minor surgical procedures y/n Management of the practice y/n Financial management y/n Professional organizations y/n Special knowledge of/interest in (the treatment of): 2 3 4 5 6 7 8 1-5 Diabetes mellitus 1-5 Astma and/or COPD 1-5 Cardiovascular diseaeses 1-5 Please indicate what percentage of patients with … disorders you manage yourself: * # 1 5 point scale7 5 point scale 8 1-5 Anxiety 1-5 Depressive symptoms 1-5 Medically unexplained symptoms 1-5 Theoretical range unless stated otherwise. Range observed. The Algorithm method) scores the first 8 items as positive if the answers are: (‘more than half the days’ or ‘nearly every day’) and as or negative if the answers are (‘“not at all’ or ‘several days’). The ninth item is scored as positive if the answer is (‘several days’ or ‘more than half of the days’ or ‘nearly every day’) and is scored negative if the answer is (‘not at all’). Five or more positive items is counted as presence of depression. The “Severity score” scores all items with 0 (‘not at all’), 1 (‘several days’), 2 (‘more than half of the days’) or 3 (‘nearly every days’). The range is from 0 to 27. To score panic we assessed the two first panic questions of the PHQ. Panic was deemed present if the first screening question was positive and 2 or more items (out of 3 items) of the second panic question were positive. We did not use the third question. To score ‘other anxiety’ we assessed the two anxiety questions of the PHQ. A patient scores positive for ‘other anxiety’ with a positive answer on the screening question (Over the last 4 weeks, how often have you been bothered by any of the following problems: feeling nervous, anxious, on edge, or worrying a lot about different things?) and 4 or more (out of six) positive answers (‘several days’ or ‘more than half of the days’) on the second question. Problems on GPs’ problem list coded with the international classification of primary care. See appendix B. Robbins JM, Kirmayer LJ, Hemami S. Latent variable models of functional somatic distress. J Nerv Ment Dis 1997; 185(10):606-615. 5 point scale: no special interest: little interest; normal interest; more than normal interest; very much interest. 5 point scale: 0%; 25%; 50%; 75%; 100% CHAPTER 8 111 Setting Data were collected in 41 practices of GPs, participants in the GP-based continuous morbidity registration network of the Department of General Practice at the Academic Medical Center of the University of Amsterdam. The practices are located in health centres in a suburban part of Amsterdam (39 practices) and in two single-handed practices in a nearby rural area. In the GP network, electronic medical record data are routinely extracted for research purposes. Study population All patients of 18 years or older enlisted with the GPs between January 1, 2008 and January 1, 2010, were potentially eligible for this study. Patients were classified based on the number of consultations with the GP (consultations in the surgery and house calls). Consultations with other practice staff were excluded because these contacts are mostly initiated by the GP or his/her staff and very often involve planned monitoring of chronic diseases. Patients who according to their GP were unable to participate because of severe mental disease, mental retardation or illiteracy (literate in neither Dutch nor English) were excluded. Frequent attenders were defined as those patients whose attendance rate ranked in the top 10th centile of four age groups (18-30; 3145; 46-60; 61+ years), separately for men and women.12;13 Frequent attendance was determined for the years 2008 through 2011. Incident FA during one year (1yFAs) was defined as attendance in the top 10th centile during 2009, but not in 2008. 112 Frequent attendance was defined as 2yFA if 1yFAs were FA in one of the consecutive years (2010 or 2011) or as persistent FAs (pFAs) if 1yFAs were FA both in 2010 and 2011. Follow-up procedure From the list of eligible 1yFAs (FA in 2009, but not in 2008), GPs excluded those people who were unable to participate because of severe mental disease, mental retardation or illiteracy. Subsequently, an invitation letter signed by their own GP was sent to the selected patients. The letter describing the study also contained an informed consent form and the baseline questionnaire. In case of no response within two weeks, a reminder was sent. All patients who returned the informed consent were enrolled and received a follow-up questionnaire after one and two years. Data quality The patient questionnaires were predesigned to enable scanning of the results electronically (Saxion Market Research; Post box 70.000, 7500 KB Enschede, the Netherlands). Because patients with missing data were telephoned or emailed within 10 days to achieve optimal completeness of data, we had little missing data and saw no need to for multiple imputation. Outcome measure Persistence of frequent attendance during 2010 or 2011 or both was the outcome. WHY DO THEY KEEP COMING BACK? Figure 1. Flow diagram Selection in 41 general practices: Total number of enlisted patients • total Na = 44,700 • > 17 year: 34,899 Eligible Frequent Attenders • N = 2,311 (FAb in 2009, not in 2008) Exclusion by the GPc N = 132 Invited for participation N= 2,179 No response = 1,433 Response N = 746 Refusal = 123 Participants T0 N = 623 d • • Death = 6 Moved house = 8 • • Death = 8 Moved house = 8 Participants T1 N = 609 ; response 526 Participants T2 N = 593 (623-30) Response = 511 Patients analysede: N= 497 a b c d e Number Frequent Attender General Practitioner All with written consent Patients with known frequent attender status in all 3 years CHAPTER 8 113 were measured using the Patient Health Questionnaire (PHQ).31 The Potential psychosocial aetiological factors The following instruments were used to capture the potential aetiological factors for persistence of FA: 1. 2. Somatoform determinants: The Illness Attitude Scales (IAS) measures fears, attitudes and beliefs associated with health anxiety and illness behaviour.21-26 The validated subscale for ‘health anxiety’ consists of eleven items on five-point scales.23;24 The validated subscale ‘illness behaviour’ has six items on the use of healthcare and the impact of physical complaints on work, concentration and leisure. The four steps of the scales are labelled from ‘no’ to ‘most of the time’. Because the IAS subscale ‘illness behaviour’ also measures use of healthcare, which is our outcome, we reanalysed our data after omitting the healthcare use-related questions from the IAS to avoid circular reasoning. Locus of control (“mastery”) was assessed by the 7-item PaerlinSchooler Mastery Scale, adapted version.21;27-29 This scale measures the extent to which one feels to be in control over changes in one’s own life (locus of control). Items are answered on a 5-point scale ranging from ‘strongly disagree’ to ‘strongly agree’. 3. Life events were assessed via the Life Event Questionnaire (LEQ) to measure a patient’s total number of negative life events in the previous 3 and 12 months.30 4. Depression and anxiety disorders 114 PHQ has been validated in FAs.32-34 We used the algorithm score to assess depression (assessing the 9 items of the Diagnostic and Statistical Manual of Mental Disorders IV text revision, DSM-IV-TR). To assess panic disorder we used the two panic questions and to assess ‘other anxiety’ the two other anxiety questions of the PHQ. See appendix 6 for the above mentioned instruments. Other (confounding) factors We considered a large set of potential confounders: • Demographic factors • (Somatic) problems as registered by the GP on the problem list in the electronic medical record (See the box). For the definition of a medical problem we refer to our previous work.1 • And GP characteristics and work style were measured using a questionnaire (see appendix 3). See the box for more details. Statistical analysis Multilevel ordinal multiple logistic regression analysis with GPs as a random intercept was performed with persistence of frequent attendance during zero, one or two of the following years as the ordinal outcome variable. We used a proportional odds model combining two analyses: 1yFA versus (2yFA and 3yFA) and (1yFA and 2yFA) versus 3yFA to show the associations WHY DO THEY KEEP COMING BACK? Table 1. Characteristics of Frequent Attenders who gave Informed Consent and who refused Participation. a b c Informed consent Refused participation p-value Na 623 1556 Age (mean, SDb) 52 (17) 45 (18) 0.00 Female, % 52.7 51.4 0.00 N of contacts with GP in 2009 (mean, SD) 9.1 (3.6) 8.2 (3.7) 0.00 N of problems on problem list(mean, SD) 2.7 (2.7) 2.2 (2.3) 0.00 Psychological problemsc (%, SD) 7.7 (20) 8.1 (20) 0.68 N indicates number SD indicates standard deviation Psychological problems as percentage of all problems on the problem list of General Practitioners’ characteristics and persistence of frequent attendance (Table 3) and the associations between our main variables and persistence of frequent attendance (Table 4).The ordinal logistic model assumes that a predictor’s effect on the probability of zero years frequent attendance versus one or two years is the same as that for zero and 1 year versus two years. Therefore, a single odds ratio expresses the effect of each predictor. We measured illness attitude and lack of mastery only at baseline and all other variables at baseline and after one and two years. We estimated the effect of psychosocial factors while controlling for demographic factors, somatic diseases and GP characteristics. We also applied a simpler model, using only the baseline values of the aetiological factors. We tested the proportional odds assumption using the parallel lines test. We assumed for all covariables in the model that the difference between the log odds of being a 3yFA and a 2yFA, respectively is the same as the difference of the log odds of being a 2yFA and 1yFA, respectively. A priori defined interaction terms between somatic, psychological and social factors were considered using a binary logistic model with 2yFA (versus 0 and 1yFA) as the dependent variable. SPSS 20.0 for windows was used for the analyses. We report odds ratios and their corresponding 95% confidence intervals. Ethics approval The study was conducted according to the Dutch legislation on data protection (Ministry of Justice, the Netherlands). Ethics approval was waived by the Medical Ethics Committee of the Academic Medical Center of the University of Amsterdam. All participants gave their written informed consent. CHAPTER 8 115 Table 2. Characteristics of 1 year, 2 year and 3 year Frequent Attenders at Baseline Frequent attender status# 1yFAs* 2yFAs* 3yFAs* Number 307 155 35 Age (mean,SD) 52 (15) 54 (18) 55 (14) 0.78 Female 52 51 49 0.88 Education: none or low 20 24 31 0.30 p-value Paid work or study 61 48 41 0.009 Living alone or with children 30 37 38 0.23 Ethnicity 0,85 - Dutch % 83 79 85 - Surinamese % 7 9 6 Class of working hours (mean,SD) 3.64 (1.04) 3.74 (1.02) 3.80 (1.15) 0.74 Body mass index (mean,SD) 26.4 (5.0) 27.3 (5.2) 28.7 (5.8) 0.41 Mastery score (mean,SD) 18.89(5.27) 16.99(5.54) 16.15(6.26) 0.33 - Health anxiety (mean,SD) 11.15(7.97) 12.49(8.17) 13.67 (9.12) 0.55 - Illness behaviour (mean,SD) 8.95(4.43) 10.83(4.69) 12.82(4.73) 0.032 Number of life eventsb (mean,SD) 2.30 (2.38) 2.68 (2.37) 2.60(2.48) 0.68 4.08 (4.71) 6.07 (5.83) 8.61(6.08) 0.017 5 10 18 <0.001 a Illness attitude scale (IAS)b Depression score PHQ (mean,SD) c; b Depression (PHQ) c Panic complaints (PHQ) 4 7 17 0.013 Other anxiety (PHQ) 7 14 17 0.017 - Total number of problems (mean,SD) 1.57(1.54) 1.86(1.77) 2.34(1.97) 0.18 - Any cardiovascular disease 39 36 27 0.76 - Any cancer diagnosis 10 7 6 0.62 - Any respiratory disease 17 21 26 0.33 - Any addiction 6 4 0 0.34 - Any social problem 2 3 3 0.69 - Any medically unexplained symptoms 9 13 26 0.012 - Any psychological problem 8 7 9 0.86 - Any digestive tract problem 13 15 26 0.15 - Any locomotor tract diagnosis 15 23 37 0.003 - Any skin disease 9 10 11 0.83 Problems on GP’s Problem listd Legend on page 117 116 WHY DO THEY KEEP COMING BACK? Legend of table two, on page 116 a Number of working hours in 5 classes:1 (1 – 10 hours), 2 (11 – 20 hours), 3 (21 – 30 hours), 4 (31 – 40 hours) and 5 (more than 40 hours) b PHQ means Patient Health Questionnaire c We used the algorithm score to assess depression (assessing the 9 items of the Diagnostic and Statistical Manual of Mental Disorders IV text revision, DSM-IV-TR). d Problems registered by the General Practitioner in the electronic medical file (definition of a problem in previous work 1) # 1yFAs indicates frequent attenders (FAs) in 2009, but not in 2010 and 2011; 2yFAs indicates FAs in 2009 and 2010 or 2009 and 2011; 3yFAs indicates FAs in 2009, 2010 and 2011 * Numbers are percentages unless stated otherwise † We used the Chi-square test or a one-way-Kruskal-Wallis ANOVA test depending whether the variable was categorical or quantitative. Results General practitioners’ characteristics The cohort of frequent attenders The average practice size of the 41 participating GPs was 1,419 enlisted patients (range, 849-2,792) and 82% of GPs indicated their participation in academic medical education or research. After correction for patient characteristics, intra-class correlations for general practices were small indicating hardly any clustering within GPs. The GP characteristics significantly associated with persistent frequent attendance were special interest in diabetes mellitus (OR 1.40; 95%CI from 1.02 to 1.91; per point on a 5-point Likert scale.) and interest in performing minor surgical procedures (yes/no; OR 1.48; 1.05 to 2.08). See Table 3. Our sampling frame consisted of 44,700 patients who were enlisted with the participating GPs of whom 34,899 were 18 years or older. Of these 2,311 patients fulfilled the criteria for incident FA. GPs excluded 132 patients for inclusion because of illiteracy, severe mental health problems or mental retardation. Of the 2,179 patients eligible for participation, 746 (34%) returned the questionnaire and of these 623 (29%) gave informed consent (figure 1). Compared to non-consenters, consenting individuals were older, more often female, had contacted the GP more often in 2009 and had more problems on the problem list. (See Table 1) Characteristics of the cohort at baseline and after one, and two years Table 2 shows the characteristics of the cohort at baseline. Persistent FAs were less likely to be in paid work and scored higher on illness behaviour, depression, panic disorder and other anxiety. Finally they had more medically unexplained symptoms and locomotor problems on their problem lists (GPs’ patients file). Psychosocial aetiology of persistent frequent attendance We performed an ordinal logistic regression with remaining a frequent attender during zero, one or, two years of follow-up as the outcome. See Table 4. Measured at baseline, panic disorder (OR 5.40; 1.67 to 17.48), other anxiety (OR 2.78; 1.04 to 7.46), illness behaviour (per additional point on the scale; OR 1.13; 1.05 to 1.20) and lack of mastery (per CHAPTER 8 117 Table 3. Associations of General Practitioners Characteristics and persistence of frequent attendance* 95%-CIc p-valued 1.03 0.75-1.41 0.87 50 (8) 1.00 0.98-1.02 0.84 17 (9) 1.00 0.98-1.02 0.69 Anxiety 2.85 (0.77) 1.07 0.84-1.37 0.56 Depression 3.05 (0.71) 1.18 0.89-1.54 0.24 Medically unexplained symptoms 3.65 (0.70) 0.99 0.72-1.36 0.96 Diabetes 3.18 (0.64) 1.40 1.02-1.91 0.04 Respiratory diseases 3.13 (0.72) 1.04 0.82-1.33 0.72 Cardiovascular diseases 3.35 (0.53) 1.22 0.85-1.75 0.29 1.04-2.08 0.03 number/mean (%/SDa) OR Female gender 23 (56%) Age Years of experience b Special interest in managinge Special interest in a specific part of General Practice (yes/no) f Minor surgical procedures, n/%. a b c d e 16 (39) 1.48 SD: standard Deviation Odds Ratio 95% confidence interval probability value Five levels of interest (from 1 (no special interest) to 5 (very much interest)) and 5 levels of percentage of patients treated by the GP (0%-100%). Special interest (Y/N) in paedriatic medicine, geriatric medicine and management/financial aspects of care were not significantly correlated. Based on a multilevel ordinal regression analysis with two levels (GP and the pa tient) with persistence of frequent attendance during 2010 or 2011 or both as the outcome. The Akaike Information Criterion (AIC) of ordinary logistic model was 1086.80, the AIC of ramdom effects logistic regression model 1084.03 (p-value 0.029). f * additional point on the scale; OR 1.08; 1.01 to 1.15) increased persistence of frequent attendance. Measured over all 3 years, other anxiety (OR 2.0; 1.29 to 3.10) and the number of life events in 3 years (OR 1.06; 1.01-1.10 per event; range of 0 to 12), increased the chance of persistence of frequent attendance. The analysis showed a 5.6 percent increase in the odds of remaining a frequent attender for every additional negative life event (OR 1.06; 1.01 to 1.10). 118 This means that for a patient with for example three negative life events in 3 years, the odds of repeated frequent attendance was 18 percent higher than for a similar person without negative life events ((1.056)3). Psychosocial patient variables of GPs’ electronic records were not significantly related to repeated frequent attendance. We tested the parallel lines assumption underlying the ordinal model and found that for all variables, except the total WHY DO THEY KEEP COMING BACK? addictive problems summed over three years, the p-values were > 0.52. Overall, we interpreted this finding as supportive for the more parsimonious model whose parallel lines test p-value was 0.78. Additional effect on persistence of frequent attendance of (combinations of ) psychological, social and somatic problems The probabilities of becoming a pFA were 0.16 (95%CI from 0.06 to 0.25), 0.12 (0.03 to 0.21) and 0.13 (0.06 to 0.19) for presence of any somatic, any psychological and any social problem, respectively. See figure 2. However, we found no evidence that the effects (odds ratios) of combinations of these factors were larger than those predicted on the basis of multiplying the odds ratios of separate effects (p-values for interactions > 0.25; logistic models tests interactions on a multiplicative scale). Discussion In a cohort of incident frequent attenders prospectively followed during two years, we found that presence of a panic disorder, presence of other anxiety, the occurrence of negative life events in the previous year (all measured at baseline and over all three years), illness behaviour and lack of mastery (both measured at baseline) were associated with persistent frequent attendance during one or two of the following two years. Presence of a combination of chronic somatic disease with social and/or psychological problems gave no rise to effects over and above what might be expected based on their separate effects. We found no strong evidence of effects of GP characteristics. Strengths of this study The prospective design with the clear focus on psychosocial determinants provides a good insight into aspects of the psychosocial aetiology of persistence of frequent attendance. The large variability in FAs included in this study yielded ample analytical contrast to robustly estimate associations of many of the determinants with persistence of frequent attendance.35 For example, the number of life events varied between 0 and 12 and the mastery scale from 5 to 26 (Table 2).36 The design with three annual questionnaires and data from GPs’ patient files provided us with a rich database. The tests for psychological problems have been specifically validated in FAs. We also tested for an enhancing effect of combinations of somatic, psychological and social determinants but found none. We achieved good data quality without imputation of missing data by automated scanning of predesigned questionnaires and a rigorous phoning and mailing approach of patients in case of missing data. We also used morbidity prevalence data of a GP database in which GPs register medical problems over a long period of time. As most GPs receive regular feedback on their registration activities, these registration data are of good quality, especially for somatic and psychological problems. 37;38 As GPs register these problems during consultations, the quality of this registration may be better for frequent attenders who have many consultations. CHAPTER 8 119 Table 4. Results from a multilevel ordinal logistic regression model with 0, 1 or 2 years as a frequent attender during follow-up1 Results of the Patient Questionnaire OR* 95% CI* P-value # 1.06 1.01-1.11 0.02 Total over 3 years2 Life events during last 12 months Major depression 1.22 0.91-1.62 0.19 Other anxiety 2.00 1.29-3.10 0.002 Panic disorder 1.63 0.89-2.99 0.11 Life events during last 12 months3 1.09 0.95-1.24 0.21 3 1.08 1.01-1.15 0.02 Measured at baseline Lack of mastery Major depression 0.62 0.18-2.09 0.44 Other anxiety 2.78 1.04-7.46 0.04 5.40 1.67-17.48 0.01 Health anxiety 1.04 1.00-1.08 0.07 Illness behaviour3 1.13 1.05-1.20 <0.001 Illness behaviour without use of healthcare 4 1.14 1.03-1.26 0.01 Any addictive behaviour 0.68 0.37-1.23 0.20 Any social problem 1.25 0.53-2.97 0.61 Panic disorder 3 Results from the Electronic Medical Record Total over 3 years Any medically unexplained symptom 0.81 0.53-1.24 0.34 Any other psychological problem 0.89 0.59-1.36 0.60 Any addictive behaviour 0.17 0.02-1.79 0.14 Any social problem 1.84 0.21-16.08 0.58 Any medically unexplained symptom 0.93 0.26-3.33 0.91 Any other psychological problem 0.44 0.12-1.65 Measured at baseline 1. 2. 3. 4. * # Based on a multilevel ordinal regression analysis with two levels (GP and the patient). Persistence of frequent attendance during 2010 or 2011 or both was the outcome. Each of the variables was modelled separately after correcting for the set of confounders: age, gender, education, ethnicity, living situation, work, body mass index, total number of problems, Chronic Obstructive Pulmonary Disease/asthma, chronic vascular diseases, cancer, skin, locomotor tract disorders and digestive tract disorders. The sum or the mean of the values of a predictor across the three time points. Per extra life event or extra point on the used scales. Ilness behaviour without questions about use of healthcare but including questions about impact on work, concentration and leisure. OR indicates Odds Ratio; CI indicates Confidence Interval. The p-values are Wald tests (estimated parameters / standard error). 120 WHY DO THEY KEEP COMING BACK? proportion of pFA patients Figure 2. The probability of persistence of frequent attendance in 2010 and 2011 (pFA) in primary care patients who attended frequently in 2009 (and not in 2008) versus all other cohort patients depending on the presence of any (combination of) chronic somatic, psychological or social problems at baseline.1 0,35 0,30 0,25 0,20 0,15 00,10 0,05 P P at ic al + So m So ci So og ic Ps al + yc h ol So og ci ic al + al + ic at m So al ic og ol Ps yc h ol Ps yc h ci al ic at + yc h So m So ol P P P ci ic og at m Ps So al al P ic P( #) no P 0,00 1 2 3 4 # Probability for pFA 2 95%CI3 SE4 Number of P # 0.06 0.00-0.12 0.03 Any somatic P 0.16 0.06-0.25 0.05 Any psychological P 0.12 0.03-0.21 0.05 Any social problem 0.13 0.06-0.19 0.03 somatic+psychological P 0.25 0.13-0.36 0.06 somatic+social P 0.20 0.12-0.29 0.04 psychological+social P 0.23 0.17-0.28 0.03 somatic+psychological+social P 0.29 0.25-0.34 0.02 The presence of problems was taken from the electronic medical records of the general practitioners. Bars indicate proportions; antennas are standard errors; pFA is persistent frequent attender. pFA means persistent frequent attendance, frequent attendance during 20092011 CI mens Confidence Interval SE means standard error P means Problems; probability per extra problem CHAPTER 8 121 Limitations of this study We see the following limitations. First, we used a proportional definition of FA. Although in our opinion this definition makes sense, its arbitrary cut-off of 90% has evident disadvantages as some patients may drop just below the cut-off (89th centile e.g.) the next year. However, definitions based on some absolute number of visits share this limitation. Second, registration in GP practices has its limitations.37-39 Some registered problems could be overreported if resolved problems are not removed, in recurrent (for instance housing problem) or temporary diseases (for instance a depressive episode). This could lead to overestimation of their prevalence and to bias in the estimations of the effect of psychological and social factors on persistence of frequent attendance.40 Fourth, the participating GPs appeared relatively similar on some potential risk factors such as interest in psychosocial diagnosis and treatment: Eighty-two percent of the GPs were involved in educating medical students, vocational training of future GPs and/or research activities. Also, GPs might have tended to answer positively when asked about their special interests in medical issues and treatments. This may lead to underestimation of the effects of GP characteristics, if any. The association of interest in surgical procedures and diabetes with persistence of frequent attendance may be explained by more follow-up consultations in patients with these problems. Fifth, although we collected many patient characteristics and clinical data, there may of course be undocumented determinants of pFA. For 122 example, in this study we had no data on perceived health status and quality of life, which are both inversely associated with persistence of frequent attendance.10 Sixth, this study originates in the Dutch healthcare system with a strong and well-organized primary care in which most costs are covered by insurances and GPs try to provide continuity of care and integrated care (somatic and psychosocial care). This specific context may restrict the generalisability of our results to countries with a comparable healthcare system such as, for example, the United Kingdom or Health Maintenance Organizations in the United States.41 For reasons of feasibility, we chose well known, validated, but easy to use questionnaires and not diagnostic interviews to assess depression, panic disorder (PHQ) and somatoform disorders (IAS). This may have resulted in some misclassification of depression, anxiety and somatoform disorders.23;32;34 Finally, about two thirds of the eligible 1yFAs refused to participate in our study. Because we focus on aetiology and ample analytical contrast, representativeness is not the issue and we do not think this influenced our results negatively.40 We analysed a cohort of new FAs and our results are therefore limited to frequent attendance during 3 years and we cannot determine whether our results are applicable to frequent attendance of longer duration. Relevant literature The literature about the causes of persistent FA is inconsistent and its straightforward interpretation is hampered by methodological differences (aetiological and causal versus predictive WHY DO THEY KEEP COMING BACK? noncausal outlooks or confusion about these two) and different definitions of frequent attendance (proportional versus fixed cutoff point). In prospective cohort studies, using a proportional definition, low physical quality of life, low educational level and psychological distress (Hopkins Symptom Check List and Whiteley-7) predicted persistence of frequent attendance in the next two consecutive years.7;10 Using a fixed cutoff definition of FA, one study found that female gender, obesity, former frequent attendance, fear of death, alcohol abstinence, low satisfaction, and irritable bowel syndrome were risk factors for persistence of frequent attendance during at least three out of the four later years.42 Another study concluded that the Ambulatory Diagnosis Groups “unstable chronic medical conditions”, “see and reassure conditions”, “minor time-limited psychosocial conditions”, and “minor signs and symptoms” predicted persistence of frequent primary care use the next year.15 We explicitly chose a proportional definition of frequent attendance per age and sex group.12;13;43 This hampers a comparison with the results of studies that chose fixed numbers of consultations to select FAs.15;42 We confirmed the association of psychological distress and pFA.7 In the Eighties some authors postulated that inadequate interpersonal dynamics between patients and their GP could cause more, inappropriate and unnecessary consultations, testing and treatments. These authors also emphasized that families tend to be consistent in illness and consultation patterns over the years and generations.16-18;44-46 However, in our study we found little evidence of associations between GP characteristics and persistence of FA. This may suggest that patient determinants are more important in explaining persistence of frequent attendance than GP characteristics, although a larger and more diverse group of GPs and more detailed information about the GPs may be needed to corroborate this. In our study about healthcare expenditures of FAs we hypothesized that the higher costs of FAs in primary and secondary care could be explained by not yet diagnosed medical problems.20 The results of this prospective cohort shows that undiagnosed anxiety and somatoform disorders, life events and poor mastery may partly explain this frequent use of (primary) healthcare by persistent FAs. Clinical and public health implications Frequent attenders of primary care and persistent FAs in particular, have more somatic, social and psychological problems and incur higher costs than non-FAs. 1;20 Our results imply that in 1yFAs, repeated FA is increased by anxiety, illness behaviour (also after omission from the IAS scale of the questions about use of healthcare), negative life events and low mastery and that, if our findings are valid, a proportion of pFA is preventable by eliminating these causes.47-51 Thus, adequate detection and treatment of these conditions in FAs may result in a better quality of life of these patients and a decrease in attendance and costs. As most somatic problems in this patient group are already dealt with relatively effectively CHAPTER 8 123 in usual GP care, the largest effects are likely to be gained by care (programmes) which address psychological problems (panic, other anxiety, depression) of the involved patients and which try to strengthen the self-efficacy of patients (mastery).52 Although treatment of anxiety and depression has been shown to be effective 53-59, the effect of this treatment in preventing persistence of FAs is unclear and needs to be demonstrated in research.60 anxiety disorders, illness behaviour, negative life events and poor mastery over one’s life were found to be associated with persistence of frequent attendance after adjustment for a large set of confounders. GPs appeared to contribute little to persistence of frequent attendance, although participating GPs may have been too similar to detect potential features of importance. In light of the budget problems in many healthcare systems and the high costs of (persisting) FAs not explained by known morbidity, we think it might be beneficial for these patients and might reduce expenditures to develop and test interventions to diminish persistence of frequent attendance.6;60 We thank the GPs involved in the Network of General Practitioners of the Academic Medical Centre-University of Amsterdam (HAG-net-AMC) for their continuous efforts to keep the electronic medical records updated and their support during this study and Alice Karsten, Gerda van Zoen and Nienke Buwalda for their logistical support. Acknowledgments Implications for further research More incident FAs, using psychiatric interviews and measures of quality of life are needed to provide more insight in the difference between PHQ diagnoses and diagnoses after a Structured Clinical Interview for DSM Disorders (SCID diagnosis) and the role of Quality of life measures. Subsequently, randomised trials may determine the extent to which interventions aimed at modifying anxiety disorders, illness behaviour and mastery in this specific group of patients improve quality of life and reduce attendance and costs of (persistent) frequent attenders. We’d like to give a special tribute for our deceased colleague, Leo Beem, for his helpful statistical work at the beginning of this project. Conflicts of interests: The authors have no competing interests to report. Funding: This study was financed by a grant from the Netherlands Organisation for Health Research and Development (ZonMw); Alledaagse ziekten no. 42011002. Conclusion In patients who frequently attended their general practitioners for one year, panic, 124 WHY DO THEY KEEP COMING BACK? References (1) Smits FT, Brouwer HJ, ter Riet G., van Weert HC. Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC Public Health 2009; 9(1):36. (10) Rifel J, Svab I, Selic P, Rotar Pavlic D, Nazareth I, Car J. Association of Common Mental Disorders and Quality of Life with the Frequency of Attendance in Slovenian Family Medicine Practices: Longitudinal Study. PLoS ONE 2013; 8(1):e54241. (2) Ward AM, Underwood P, Fatovich B, Wood A. Stability of attendance in general practice. Fam Pract 1994; 11(4):431-437. (11) Koskela TH, Ryynanen OP, Soini EJ. Risk factors for persistent frequent use of the primary health care services among frequent attenders: a Bayesian approach. Scand J Prim Health Care 2010; 28(1):55-61. (3) Botica MV, Kovacic L, Tiljak MK, Katic M, Botica I, Rapic M et al. Frequent attenders in family practice in Croatia: Retrospective study. Croatian Medical Journal 2004; 45(5):620-624. (4) Carney TA, Guy S, Jeffrey G. Frequent attenders in general practice: a retrospective 20-year follow-up study. Br J Gen Pract 2001; 51(468):567-569. (5) Andersson S-O, Lynoe N, Hallgren C-G, Nilsson M. Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scandinavian Journal of Primary Health Care 2004; 22(2):91-94. (6) Smits FT, Brouwer HJ, Zwinderman AH, Mohrs J, Smeets HM, Bosmans JE et al. Morbidity and doctor characteristics only partly explain the substantial healthcare expenditures of frequent attenders: a record linkage study between patient data and reimbursements data. BMC Fam Pract 2013; 2013(1):138. (7) Vedsted P, Fink P, Olesen F, MunkJorgensen P. Psychological distress as a predictor of frequent attendance in family practice: a cohort study. Psychosomatics 2001; 42(5):416-422. (8) Tomenson B, McBeth J, Chew-Graham CA, Macfarlane G, Davies I, Jackson J et al. Somatization and health anxiety as predictors of health care use. Psychosom Med 2012; 2012(6):656-664. (9) Vedsted P, Fink P, Olesen F, MunkJorgensen P. Psychological distress as a predictor of frequent attendance in family practice: A cohort study. Psychosomatics: Journal of Consultation Liaison Psychiatry /9; 42(5):416-422. (12) Vedsted P, Christensen MB. Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health 2005; 119(2):118-137. (13) Smits FT, Mohrs J, Beem E, Bindels PJ, van Weert HC. Defining frequent attendance in general practice. BMC Fam Pract 2008; 9(1):21. (14) McDaniel SH, Campbell T, Seaburn D, van EJ, Grol R, Huygen F et al. Treating Somatic Fixation: A Biopsychosocial Approach: When patients express emotions with physical symptoms. Soc Sci Med 1982; 1982(13):717-725. (15) Naessens JM, Baird MA, Van Houten HK, Vanness DJ, Campbell CR. Predicting persistently high primary care use. Ann Fam Med 2005(4):324-330. (16) Rosendal M, Fink P, Bro F, Olesen F. 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Psychother Psychosom 2008;77(6):337-350. (22) Kellner R, Abbott P, Winslow WW, Pathak D. Fears, beliefs, and attitudes in DSM-III hypochondriasis. J Nerv Ment Dis 1987(1):20-25. (23) Speckens AE, Spinhoven P, Sloekers PP, Bolk JH, van Hemert AM. Illness Attitudes Scale dimensions and their associations with anxiety-related constructs in a nonclinical sample. J Psychosom Res 1996; 1996(1):95-104. (24) Speckens AEM, Spinhoven P, Sloekers PPA, Bolk JH, van Hemert AM. A validation study of the Whitely Index, the Illness Attitude Scales, and the Somatosensory Amplification Scale in general medical and general practice patients. J Psychosom Res 1996; 40(1):95-104. (31) Spitzer RL, Kroenke K, Williams JB. Validation and utility of a self-report version of PRIME-MD: the PHQ primary care study. Primary Care Evaluation of Mental Disorders. Patient Health Questionnaire. JAMA 1999; 282(18):17371744. (32) Wittkampf K, van RH, Baas K, van de Hoogen H, Schene A, Bindels P et al. The accuracy of Patient Health Questionnaire-9 in detecting depression and measuring depression severity in high-risk groups in primary care. Gen Hosp Psychiatry 2009; 31(5):451-459. (33) Baas KD, Wittkampf KA, van Weert HC, Lucassen P, Huyser J, van den Hoogen H et al. Screening for depression in high-risk groups: prospective cohort study in general practice. The British Journal of Psychiatry 2009; 194(5):399403. (34) Wittkampf KA, Baas KD, van Weert HC, Lucassen P, Schene AH. The psychometric properties of the panic disorder module of the Patient Health Questionnaire (PHQ-PD) in high-risk groups in primary care. J Affect Disord 2010:Nov. (35) Miettinen O.S. Theoretical Epidemiology: principles of occurrence research in medicine. New York: Wiley; 1985. (25) Kellner R. Hypochondriasis and somatization. JAMA 1987; 1987(19):27182722. (36) Rothman K, Greenland S. Modern Epidemiology. Philadelphia: LippincottRaven Publishers; 1998. (26) Sirri L, Fava GA, Sonino N. The Unifying Concept of Illness Behavior. 2013. (37) Brouwer HJ, Bindels PJ, van Weert HC. Data quality improvement in general practice. Fam Pract 2006; 23(5):529536. (27) Pearlin LI, Schooler C. The structure of coping. J Health Soc Behav 1978(1):2-21. (28) Pearlin LI, Lieberman MA, Menaghan EG, Mullan JT. The stress process. J Health Soc Behav 1981(4):337-356. (29) Moser DK, Dracup K. Psychosocial recovery from a cardiac event: the influence of perceived control. Heart Lung 1995(4):273-280. (30) Saxe LL, Abramson LY. The life events scale, reliability an validity. Unpublished manuscript.; 1987. 126 (38) van den Dungen CHN, Boshuizen HC, van den Akker M, Biermans MC, van Boven K., Brouwer HJ et al. What factors explain the differences in morbidity estimations among general practice registration networks in the Netherlands? A first analysis. Eur J Gen Pract. 2008;14 Suppl 1:53-62 (39) van den Dungen C, Hoeymans N, Boshuizen HC, van den Akker M, Biermans MC, van BK et al. The influence of population characteristics on variation in general practice based morbidity estimations. BMC Public Health 2011; 11:887. WHY DO THEY KEEP COMING BACK? (40) Rothman K, Greenland S, Lash T. Modern epidemiology. Philadelphia, PA (USA): Lippincott Williams & Wilkins; 2008. (41) Dunlop S, Coyte PC, McIsaac W. Socioeconomic status and the utilisation of physicians’ services: results from the Canadian National Population Health Survey. Soc Sci Med 2000; 51(1):123-133. (42) Koskela TH, Ryynanen OP, Soini EJ. Risk factors for persistent frequent use of the primary health care services among frequent attenders: a Bayesian approach. Scand J Prim Health Care 2010; 28(1):55-61. (43) Luciano JV, Fernandez A, Pinto-Meza A, Lujan L, Bellon JA, Garcia-Campayo J et al. Frequent attendance in primary care: comparison and implications of different definitions. Br J Gen Pract 2010; 60(571):49-55. (44) van Eijk J, Grol R, Huygen F, Mesker P, Mesker-Niesten J, van MG et al. [Somatic fixation. Prevention by the general practitioner. Soc Sci Med 1982; 1982(13):717-725. (45) McDaniel SH, Campbell T, Seaburn D. Treating Somatic Fixation: A Biopsychosocial Approach: When patients express emotions with physical symptoms. Can Fam Physician 1991:451456. (46) Cardol M, van den Bosch WJHM, Spreeuwenberg P, Groenewegen PP, van Dijk L, de Bakker DH. All in the Family: Headaches and Abdominal Pain as Indicators for Consultation Patterns in Families. The Annals of Family Medicine 2006; 4(6):506-511. (47) Cuijpers P, van Straten A, Schuurmans J, van Oppen P, Hollon S, Cuijpers P. Psychotherapy for chronic major depression and dysthymia: A metaanalysis. Clin Psychol Rev 2010 febr; 30(1):51-62 (48) Cuijpers P, van Straten A, Warmerdam L. Problem solving therapies for depression: A meta-analysis. European Psychiatry 2007; 22(1):9-15. (49) Reger MA, Gahm GA. A metaanalysis of the effects of Internet- and computer-based cognitive-behavioral treatments for anxiety. Journal of Clinical Psychology 65[1], 53-75. 2009. CHAPTER 8 127 (50) van der Feltz-Cornelis C, van der Feltz-Cornelis C. Effect of psychiatric consultation models in primary care. A systematic review and meta-analysis of randomized clinical trials. Journal of Psychosomatic Research 2010;68(6). (60) Smits FT, Wittkampf KA, Schene AH, Bindels PJ, van Weert HC. Interventions on frequent attenders in primary care. A systematic literature review. Scand J Prim Health Care 2008; 26(2):111-116. (51) Morriss R, Kai J, Atha C, Avery A, Bayes S, Franklin M et al. Persistent frequent attenders in primary care: costs, reasons for attendance, organisation of care and potential for cognitive behavioural therapeutic intervention. BMC Fam Pract 2012:39. (52) Samoocha D, Bruinvels DJ, Elbers NA, Anema JR, van der Beek AJ. Effectiveness of web-based interventions on patient empowerment: a systematic review and meta-analysis. J Med Internet Res 2010(2):e23. (53) Cuijpers P, van SA, van SA, Andersson G. Psychological treatment of depression in primary care: a meta-analysis. Br J Gen Pract 2009; 59(559):51-60. (54) Cuijpers P, van Straten A, Andersson G, van Oppen P. Psychotherapy for depression in adults: A meta-analysis of comparative outcome studies. Journal of Consulting and Clinical Psychology 2008; 76(6):909-922. (55) Donker T, Griffiths KM, Cuijpers P, Christensen H. Psychoeducation for depression, anxiety and psychological distress: a meta-analysis. BMC Med 2009 Dec 16;7:79 (56) Goldberg D. The “NICE Guideline” on the treatment of depression. Epidemiologia e Psichiatria Sociale Vol 15 (1), Jan -Mar 2006;-15. (57) Kendall T, Cape J, Chan M, Taylor C, Kendall T, Cape J et al. Management of generalised anxiety disorder in adults: summary of NICE guidance. BMJ 2011(4):318-327. (58) Pilling S, Mayo-Wilson E. Recognition, assessment and treatment of social anxiety disorder: summary of NICE guidance. BMJ 2013; 346. (59) National Collaborating Centre for Mental Health. Generalised Anxiety Disorder in Adults: Management in Primary, Secondary and Community Care. 2011. 128 WHY DO THEY KEEP COMING BACK? CHAPTER 8 129 chapter 9 IS SYSTEMATIC DETECTION AND TREATMENT OF DEPRESSION AND ANXIETY IN FREQUENT ATTENDERS COST-EFFECTIVE IN COMPARISON WITH USUAL CARE BY GENERAL PRACTITIONERS? A MODELLING STUDY Judith Bosmans, Frans Smits, Veerle Coupé, Aart Schene, Henk van Weert, Gerben ter Riet. Article submitted ABSTRACT Background Frequent attenders (FAs) are defined as patients with an age and sex-adjusted attendance rate ranking in the top 10 centile within a time frame of one year. Many FAs have underlying psychiatric morbidity that may not always be detected and treated by their General Practitioner (GP). Objective To evaluate if systematic detection and treatment of depression and anxiety in FAs is cost-effective in comparison with usual GP care and whether this should be initiated after 1 or 2 years of frequent attendance. Methods A Markov model was developed to simulate the course of a cohort of 10,000 one-year FAs (1yFAs) over a period of 5 years. Main outcomes were years spent as non-FA without depression or anxiety, and Quality-Adjusted Life-Years (QALYs). Several treatment scenarios were simulated with treatment effects expressed as risk reductions of 10%, 20%, and 40% compared with usual care. Usual care consisted of the natural course of a cohort of FAs in Dutch primary care around 2011. Uncertainty was estimated using Monte Carlo simulation. Results Expected increases in years spent as non-FA without depression or anxiety and QALYs were small in all treatment scenarios. Treatment of 1yFAs resulted in larger health benefits and more cost savings than treatment of 2yFAs. The 95% credibility intervals around expected cost and effect differences were wide and included no effect. Treatment was cost-effective or dominant in comparison with usual care only if treatment was assumed to result in a spillover effect on FA-ship. Conclusion Systematic detection and treatment of depression and anxiety in FAs does not seem to be cost-effective. 132 WHY DO THEY KEEP COMING BACK? Introduction In primary care, patients who consult more often than their peers have a disproportionate effect on the workload of General Practitioners (GPs).1, 2 Because frequency of attendance is strongly associated with age and sex, 3 it is recommended to define frequent attenders (FAs) as patients with an age and sexadjusted attendance rate ranking in the top 10 centile within a time frame of one year.4, 5 Such a proportional threshold allows for meaningful comparison between practices, periods, and countries. FAs in primary care are not only frequent users of GP services, but also of other healthcare services such as emergency medical care services and hospital services leading to high healthcare costs.6-8 FAs tend to have lower education levels, and more medical and psychiatric morbidity than non-frequent attenders (non-FAs).2, 6, 9 Between 80% and 90% of the FAs frequently attend their GP for 1 or 2 years only10-13. Studies have shown that patients who are a frequent attender for 2 or more consecutive years have more chronic somatic diseases, social problems, medically unexplained somatic symptoms and psychiatric problems than one-year frequent attenders.2, 14, 15 Moreover, they have a lower quality of life and lower education levels.16 Smits et al systematically reviewed the evidence on the effectiveness of interventions aimed at FAs in influencing morbidity, quality of life and healthcare utilization rates. The interventions included in their review varied greatly in content, and were not considered effective.17 More recent studies showed that more intensive interventions aimed at healthcare providers or patients resulted in reduced medical care utilization18-20, although effects were generally small (decrease in healthcare utilization 1.5 to 6 visits per year in comparison with usual care). Because many FAs have underlying psychiatric morbidity that may not always be detected or diagnosed by their GP, it has been suggested that systematic detection and treatment of underlying disorders might improve quality of life and reduce consultation rates.21, 22 We developed a Markov model to assess whether such an intervention may be cost-effective compared to usual care and whether the best moment to intervene is after 1 or 2 years of frequent attendance. To do this we extrapolated the findings from a cohort study among one-year FAs over a period of 5 years and combined this with potential treatment effects using modeling techniques. Methods Design We developed a Markov model in which the course of a cohort of primary care patients who frequently attended their GP during one year (1yFAs) in Dutch primary care (i.e. usual care) was extrapolated over a period of 5 years and linked to cost data from a large healthcare insurer in The Netherlands. Transition probabilities for the model were estimated using the 1 year follow-up from the prospective cohort study described below. CHAPTER 9 133 Table 1. Model parameters used in the Markov model:costs, utility weights and treatment effects. Costs (¤) per year Psychological treatment in primary care ¤500 nonFA ¤2400 FA duration (years) Depression Anxiety Unknown or no morbidity Successful treatment 1 ¤6300 ¤13900 ¤4500 -- 2 ¤12400 ¤4200 ¤4400 ¤4400 >2 ¤12000 ¤6300 ¤8100 ¤8100 FA NonFA Depression 0.56 {Rifel}* 0.62 {Siskind 2010}{Paulden 2009}{Sado 2009} {Siskind 2008} Anxiety 0.55 {Rifel}* 0.66 {Konig 2009} Unknown or no morbidity 0.57{Rifel}* 0.85 {Sobocki 2008}{Burstrom 2001}{Johnson 2000} Successful treatment 0.65† Utility weights Treatment effects (Risk reduction) Effect of treatment on disorder under consideration Spillover Spillover effect effect on other on FA status disorder Usual care scenario 0% 0% 0% Alternative scenarios 10% 0%/10% 0%/10% 20% 0%/20% 0%/20% 40% 0%/40% 0%/40% 100% 100% 100% Best case scenario * † Based on unpublished data of Rifel et al[1]. Assumed that successful treatment of an underlying psychiatric disorder leads to an increase in utility of 0.1. 134 WHY DO THEY KEEP COMING BACK? Figure 2. Cost-effectiveness acceptability curves for scenarios involving a risk reduction of 20% for depression and anxiety without spillover effects on FA status. 1 scenario 3 0,9 scenario 6 0,8 scenario 8 0,7 probability cost-effective 0,6 0,5 0,4 0,3 0,2 0,1 0 0 5000 10000 15000 20000 25000 30000 25000 40000 45000 50000 threshold Cohort study A three-year prospective cohort study was performed in seven primary healthcare centres in Amsterdam, The Netherlands.8 All patients of 18 years and older registered at the participating GPs in 2007, 2008, 2009 and 2010 were eligible for this cohort study. Patients were classified according to their frequent attendance status based on information in the GP’s electronic medical record system. FAs were defined as those patients whose attendance rate ranked in the top 10th centile of four age groups (18-30 years; 31-45 years; 46-60 years; 61 years+) for men and women separately. Frequent attendance was determined for each of the years 2007, 2008, 2009 en 2010. Patients could frequently attend during 1 year (1-year frequent attenders, 1yFA), 2 years (2-year frequent attenders, 2yFA) or 3 years or more (persistent frequent attenders, pFA). Patients who were not a frequent attender in any of these years were classified as non-frequent attenders (non-FAs). Underlying morbidity The presence of depression and anxiety (panic disorder and other anxiety) was assessed using the Patient Health Questionnaire (PHQ) at the beginning of 2010 and again after 1 year.23 We used the algorithm score to assess depression (assessing the 9 items of the DSM-IVTR). To assess panic disorder we used the two panic questions and to assess CHAPTER 9 135 ‘other anxiety’ the other two anxiety questions of the PHQ. Patients who were both depressed and anxious according to the PHQ were classified as being depressed. Patients who were neither depressed nor anxious according to the PHQ were classified as having unknown or no morbidity. Patients in this state may have no morbidity or another underlying different FA states. disorder, such as a somatization disorder, that might (partly) explain the FA status of these patients, but whose exact nature was unknown to us. no or unknown morbidity. Cost data To obtain realistic data on costs of primary and secondary care, and prescription medication, data from the prospective cohort study were linked to a database with reimbursement claims from Achmea/ Agis, a large healthcare insurer in The Netherlands.24 Linking was performed using a unique and obligatory “citizen service number” by a certified trusted third party.8 Table 1 lists the cost estimates per year for the different FA states. Quality-adjusted life-years Utility weights were estimated based on available literature.16 Utility weights express the relative desirability of a health state on a scale of 0 (‘dead’) to 1 (‘perfect quality of life’). For example, a utility score of 0.56 indicates that a patient is in a health state that is valued as 56% of perfect health. Using these utility weights, Quality-Adjusted Life-Years (QALYs) were calculated by multiplying the utility weight associated with a given FA state by the number of person-years spent in that state. Table 1 lists the utility weights for the 136 Model structure In the Markov model, 1yFAs were classified into three categories: depression, anxiety, or no or unknown morbidity. Figure 1 shows a simplified model for 1yFAs with depression. The full model contains parallel chains for 1yFAs with anxiety and After one year, a 1yFA may become a 2 year frequent attender (2yFA) or a nonfrequent attender (non-FA) either with depression or anxiety, or no or unknown morbidity. A 2yFA may remain a frequent attender during the following year and, thus, become a persistent frequent attender (pFA) or go to a non-frequent attender state either with depression, anxiety or no or unknown morbidity. The model assumes that people who go from a frequent attender state to a non-frequent attender state remain in that state indefinitely. If a patient has been successfully treated for depression or anxiety, the patient remains 1 year in a state reflecting this. It was assumed that, after 1 year in this state, a patient moves to the state of being a nonFA with no or unknown morbidity. Treatment scenarios The Markov model was used to evaluate the cost-effectiveness of systematic detection and treatment of depression and anxiety in frequent attenders compared to usual care. Treatment effects were incorporated in the model as relative risk reductions. Thus, the probability of moving from a state with depression or anxiety to any other state with depression or anxiety, WHY DO THEY KEEP COMING BACK? respectively, was reduced by 10, 20 or 40 percent points. For example, using a treatment effect of 40% the probability of going from the state 1yFA+depression to 2yFA+depression is reduced from 0.10 to 0.06. Different treatment scenarios were evaluated (see Table 1). Firstly, treatment could be initiated in 1yFAs or 2yFAs. Secondly, treatment could be aimed at FAs with depression, FAs with anxiety, or both at FAs with depression and FAs with anxiety. Thirdly, spillover effects on psychiatric morbidity and frequent attender status were modeled. This allows that, for example, a treatment aimed at depression also decreases the probability to become anxious or remain a frequent attender. A best case scenario was also modeled in which risk reductions of 100% were assumed for depression, anxiety and FA status. In accordance with recent guidelines for depression and anxiety, treatment was assumed to consist of psychotherapy given by a psychologist in primary care. Based on Baas et al, we assumed that patients receive on average 6.1 treatment sessions 25 that were valued using the standard price for a psychologist (€80).26 This was rounded up to €500 per patient treated. Usual care The reference scenario was the scenario based on the transition probabilities observed in the cohort study. This can be considered the course under circumstances of usual care received by frequent attenders in general practice. Usual care in this cohort was provided in a well-serviced multi-ethnic setting in Amsterdam, The Netherlands in the years between 2007 and 2010. In the Netherlands, the GP has a gatekeeper function for care. Dutch primary care guidelines for treatment of depression recommend education and short-term psychological therapy. Only in cases of severe depression, referral and/ or antidepressants are recommended. Dutch guidelines for anxiety recommend education and short-term psychotherapy if preferred. In persistent cases, cognitive behavioural therapy or prescription of antidepressants is recommended. Grol et al showed that on average 61% of the guideline recommendations is followed in Dutch general practice.27 Cost-effectiveness analyses The cost-effectiveness of the various treatment scenarios as compared to usual care was estimated by simulating the FA trajectories of a cohort of 10,000 1yFAs over a period of 5 years with a cycle length of 1 year. For each treatment scenario, the number of years in a non-FA state with unknown or no morbidity, QALYs and costs were estimated. Costs were discounted at 4% and effects at 1.5% annually as recommended by Dutch costing guidelines.26 We calculated Incremental Cost-Effectiveness Ratios (ICERs) for each scenario in comparison with the reference scenario by dividing the difference in discounted costs between the two scenarios by the difference in discounted effects. CHAPTER 9 137 WHY DO THEY KEEP COMING BACK? 0.10 0.02 0.01 0 0.06 0.04 0.03 0 0.39 0.27 0.17 var 0 0 0 0 0.02 0.07 0.03 0* 0.12 2yFA +depression 2yFA +anxiety 2yFA +unknown 2yFA +Tx succes pFA +depression pFA +anxiety pFA +unknown pFA +Tx succes nonFA +depression nonFA +anxiety nonFA +no morbidity 0.31 0 1yFA +unknown 0 0 0 0 0 0 0.15 var 0* 0* var 0.39 0.31 0.11 0.06 0.15 0 0 0 0 0.04 0.11 0 0 0 0 0 0 0 2yFA+anxiety n=13 0 0 0 0 0 0 0 0 2yFA+Tx succes n=0 0 0.01 0 0.03 0 0 0.34 0 0.02 0 0.03 0 0 0 0 0 0 0 0 2yFA+unknown n=104 0.42 0.72 0.33 0.38 0.58 1 0.18 0 0 0 0 var 0 0 0 0 0 1yFA +anxiety 0 0 1yFA +depression 1yFA+unknown n=359 State T1 2yFA+depression n=18 1yFA+depression n=49 State T0 pFA+depression n=14 0 0 0 0 0 0 0 pFA+anxiety n=15 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0.27 0.07 0 0 0.07 0* 0 0 0 0.07 0.20 0.40 1 0.07 0.07 0.03 0 0.14 0 0.43 0.20 0.49 0 0.21 0.07 0.20 0.01 0 0 0 0 0 0 0 0 pFA+unknown n=75 Table 2. Yearly transition probabilities in a cohort study among 1 year frequent attenders. The probabilities presented in this table relate to one year frequent attenders in a usual care setting. pFA+Tx succes n=0 138 1yFA+anxiety n=45 State T0 State T1 1yFA 2yFA pFA nonFA unknown Tx success * = state at beginning of the model cycle; = state at the end of the model cycle; = 1 year frequent attender; = 2 year frequent attender; = persistent frequent attender; = non-frequent attender; = unknown morbidity other than depression or anxiety; = state after successful treatment of either depression or anxiety These transition probabilities are 0 due to the relatively small number of patients in these states. In the probabilistic sensitivity analysis, these probabilities are estimated based on a beta distribution. nonFA+ depression, n=0 0 0 0 0 0 0 0 0 0 0 0 nonFA+anxiety n=0 0.50 0 0 0 0 0 0 0 0 0 0 0 0 0 0.50 0.50 1 0* 0.50 0 0 0 0 0 0 0 0 0 0 0 0 nonFA+no morbidity, n=0 Figure 1. Simplified flowchart of the Markov model for one year frequent attenders (1yFAs) with depression. The full model contains parallel chains for 1yFAs with anxiety or unknown morbidity. 1YFA +depression 1yFA 2yFA pFA nonFA unknown or no morbidity Tx success anxiety 2YFA +depression pFA +depression NonFA +depression 2YFA +anxiety pFA +anxiety NonFA +anxiety 2YFA + unknown or no morbidity pFA + unknown or no morbidity NonFA + unknown or no morbidity 2YFA +Tx succes pFA +Tx succes 1 year frequent attender; 2 year frequent attender; persistent frequent attender; non-frequent attender; unknown morbidity other than depression or anxiety, or no morbidity; state after successful treatment of either depression or Probabilistic sensitivity analysis Probabilistic sensitivity analysis was performed by specifying beta distributions for all transition probabilities to represent uncertainty in their estimation. Next, Monte Carlo simulation was employed to randomly select values from those distributions.28 Uncertainty estimates were obtained by simulating 1000 times the course of a cohort of 10,000 1yFAs. Ninety-five percent credible intervals (95% CIs) around costs and effects were estimated based on the 2.5 and 97.5 centiles of the 1000 simulation estimates for costs and effects. Results were presented per 1,000 FAs. Cost-effectiveness acceptability curves were estimated for the scenarios involving risk reductions of 20% without spillover effects on FA status using the netbenefit approach.29 These curves show the probability that a scenario is cost-effective in comparison with the other scenarios for different thresholds for willingness-to-pay. All analyses were performed using Excel 2010. CHAPTER 9 139 Results From the prospective cohort study, data was available on frequent attender status and psychiatric morbidity (depression and anxiety) at two time points one year apart for 692 FAs. Over 2009, there were 453 1yFAs, 135 2yFAs, and 104 pFAs. Of the 1yFAs, 49 were depressed, 45 were anxious and 359 were neither depressed nor anxious, but could have unknown morbidity or no morbidity. Of the 2yFAs, 18 were depressed, 13 were anxious and 104 had other, unknown or no morbidity. For pFAs, these numbers were 14, 15 and 75 for depression, anxiety and other, unknown or no morbidity, respectively. Reference scenario The trajectories of FAs under the reference scenario are represented by the transition probabilities in Table 2 which were based on the observed transitions in the prospective cohort study. Zero indicates that these transitions are theoretically impossible, while starred zeroes indicate transitions that are possible but that did not occur in the prospective cohort. A probability of 1 indicates that patients who reach this FA state remain in this state indefinitely. Table 2 shows that after 1 year 55% of the 1yFAs with depression had become a 2yFA. For 1yFAs with anxiety and 1yFAs with no or unknown morbidity this was 33% and 21%, respectively. Of the 2yFAs with depression, anxiety and unknown morbidity, 56%, 61%, and 39%, respectively, had become a pFA after a year. Seventy-two percent, 66% and 57% of the pFAs with depression, anxiety and no or unknown morbidity, respectively, 140 had remained a pFA after a year. Thus, the longer a person resided in a FA state, the higher the probability that s/he remained in a FA state. Of the 1yFAs with depression and the 1yFAs with anxiety, 70% and 69%, respectively, did not have either of these disorders a year later. For the 2yFAs with depression and the 2yFAs with anxiety, these figures were 72% and 69%, respectively, and for the pFAs with depression and the pFAs with anxiety, 50% and 40%, respectively. Thus, in about two-thirds of 1yFAs and 2yFAs with depression or anxiety these disorders had disappeared after 1 year. Persistent FAs, however, were more likely to remain depressed or anxious. Model-based 5 year predictions for a cohort of 1,000 1yFAs under the reference scenario are shown in the first row of Table 3. On average, 4,322 out of 5000 person-years (86% of time) were expected to be spent as a non-frequent attender, 4,502 person years (90% of time) without depression or anxiety, and 4,087 personyears (82% of time) as a non-frequent attender without depression or anxiety. The number of QALYs expected over 5 years per 1,000 1yFAs was 3,927. Treatment of 1yFAs Table 3 shows estimates of the expected outcomes and costs per 1,000 1yFAs over a period of 5 years by treatment scenario. Overall, the expected effects of systematic treatment of depression and/or anxiety in 1yFAs were small and 95% credibility intervals (CrI) were wide. Even in the best case scenario that assumed that the treatment was fully effective (100% risk WHY DO THEY KEEP COMING BACK? reduction for depression, anxiety and FA status), the number of years of being a non-frequent attender without morbidity increased by only 4.5% (186 person-years, 95%CrI 38 to 334) and the number of QALYs by 1.1% (44 QALYs, 95%CrI 5 to 78) in comparison with the reference scenario. A cost reduction of 2.5% (-€330,710, 95%CrI -970,077 to 311,325) was expected under the best case scenario in comparison with the reference scenario. In general, differences in years without being a FA and without morbidity, differences in QALYs and cost differences between the depression treatment scenarios and the reference scenario were more positive than between the anxiety treatment scenarios and usual care. Table 3 shows that using the commonly accepted threshold of €20,000/QALY in The Netherlands, depression treatment scenarios with risk reductions of 10% with spillover effects on anxiety and FA status and of 20% with a spillover effect on anxiety (scenarios 4 and 6) were cost-effective in comparison with the reference scenario. This also applied for the anxiety treatment scenarios with risk reductions of 20% with spillover effects and with risk reductions of 40% (scenarios 15 to 19), and the depression and anxiety treatment scenario with risk reductions of 10% with spillover effects (scenario 21). Depression treatment scenarios assuming a treatment effect of 20% on depression and spillover effects on anxiety and FA status, and of 40% regardless of spillover effects (scenarios 7 tot 10) dominated (i.e. lower costs and larger effects) the reference scenario just as treatment scenarios for depression and anxiety with treatment effects of at least 20% with spillover effects. Figure 2 shows the cost-effectiveness acceptability curve for the scenarios 3, 6 and 8. This curve shows that when taking statistical uncertainty into account, scenario 3 is cost-effective in comparison with scenarios 6 and 8 for thresholds until 5000 €/QALY and scenario 8 for higher thresholds. However, the probabilities of cost-effectiveness do not differ greatly between scenarios. Treatment of 2yFAs Table 4 shows astimates of the expected outcomes and costs per 1,000 1yFAs after 2 years of frequent attendance by treatment scenario. Effects of treatment after 2 years of frequent attendance were smaller than treatment of 1yFAs. In the best case scenario, the expected increase in the number of person-years in a nonFA state without anxiety or depression was 1.2% (48, 95%CrI -103 to 192) and in QALYs 0.33% (13, 95%CrI -30 to 50) as compared with the reference scenario. In this scenario, cost savings in comparison with the reference scenario were 1.3% (-€175,268, 95%CrI -803,271 to 536,320). Expected cost savings and effects in comparison with the reference scenario for anxiety treatment scenarios were larger than for depression treatment scenarios. The incremental cost-effectiveness ratios in the last two columns of Table 4 show that using a cost-effectiveness threshold for QALYs of 20,000 €/QALY, only treatment scenarios assuming a risk reduction of 10% or 20% without spillover CHAPTER 9 141 Table 3. Expected costs, outcomes differences in costs and outcomes and incremental cost-effectiveness ratios in one year frequent attenders (1yFAs) per 1,000 1yFAs. FA = frequent attender; QALY = Quality-Adjusted Life-Year; Tx = treatment. 1. 2. 3. 4. 142 Rifel J, Svab I, Selic P, Rotar Pavlic D, Nazareth I, Car J: Association of common mental disorders and quality of life with the frequency of attendance in Slovenian family medicine practices: longitudinal study. PLoS One 2013, 8(1):e54241. Siskind D, Araya R, Kim J: Cost-effectiveness of improved primary care treatment of depression in women in Chile. Br J Psychiatry 2010, 197(4):291296. Paulden M, Palmer S, Hewitt C, Gilbody S: Screening for postnatal depression in primary care: cost effectiveness analysis. BMJ 2009, 339:b5203. Sado M, Knapp M, Yamauchi K, Fujisawa D, So M, Nakagawa A, Kikuchi T, Ono Y: Cost-effectiveness of combination therapy versus antidepressant therapy for management of depression in Japan. Aust N Z J Psychiatry 2009, 43(6):539-547. WHY DO THEY KEEP COMING BACK? 5. 6. 7. 8. 9. Siskind D, Baingana F, Kim J: Cost-effectiveness of group psychotherapy for depression in Uganda. J Ment Health Policy Econ 2008, 11(3):127-133. Konig HH, Born A, Heider D, Matschinger H, Heinrich S, Riedel-Heller SG, Surall D, Angermeyer MC, Roick C: Cost-effectiveness of a primary care model for anxiety disorders. Br J Psychiatry 2009, 195(4):308-317. Sobocki P, Ekman M, Ovanfors A, Khandker R, Jonsson B: The cost-utility of maintenance treatment with venlafaxine in patients with recurrent major depressive disorder. Int J Clin Pract 2008, 62(4):623-632. Burstrom K, Johannesson M, Diderichsen F: Swedish population health-related quality of life results using the EQ-5D. Qual Life Res 2001, 10(7):621-635. Johnson JA, Pickard AS: Comparison of the EQ-5D and SF-12 health surveys in a general population survey in Alberta, Canada. Med Care 2000, 38(1):115-121. CHAPTER 9 143 Table 4. Expected costs, outcomes, differences in costs and outcomes and incremental cost-effectiveness ratios of treatment in two year frequent attenders (2yFAs) per 1,000 1yFAs. FA = frequent attender; QALY = Quality-Adjusted Life-Year; Tx = treatment. 144 WHY DO THEY KEEP COMING BACK? CHAPTER 9 145 effects (scenarios 2, 3, 5 and 11) were not cost-effective as compared to the reference scenario. Using this threshold, all other scenarios were either cost-effective in comparison with the reference scenario (scenarios 4, 6, 8, 12, 14, 17, 20 and 22) or dominated the reference scenario (scenarios 7, 9, 10, 13, 15, 16, 18, 19, 21 and 23 to 26). Discussion Summary of main findings Using a Markov model, we evaluated the cost-effectiveness of systematic detection and treatment of depression and anxiety after 1 or 2 years of frequent attendance compared with usual care as provided in a multi-ethnic setting in Amsterdam. In all scenarios, 95%CrIs around expected cost and effect differences were wide and included the possibility of no difference. Treatment of 1yFAs with depression or anxiety led to larger cost savings and health benefits than treatment of 2yFAs. Assuming of spillover effects on FA status and psychiatric morbidity had a substantial influence on ICERs. Scenarios including such spillover effects were more often cost-effective compared to the reference scenario (using a threshold of 20,000 €/QALY) and more often dominated the reference scenario than scenarios excluding these effects. Comparison with existing literature In the prospective cohort study our model was based on, approximately two-thirds of the FAs had recovered from anxiety or depression after 1 year. This is high 146 in comparison with recovery rates of 50% under usual care reported in the literature.30, 31 It may be that the frequent attenders in the prospective cohort study our model was based on already received some form of care for their underlying morbidity which may partly explain their increased healthcare utilization rates. Earlier studies showed no or only minimal effects of psychological treatments aimed at undiagnosed psychiatric morbidity in frequent attenders.17 More recent studies suggest that interventions aimed at primary care providers or cognitive interventions aimed at somatising patients reduced medical care utilization, although effects were generally small.18-20 Our model shows that, against a background of a well-serviced multi-ethnic setting, even when treatments are considered to be fully effective, the benefits in terms of costs and effects are likely to be small. It has been suggested that FAs have unrecognized morbidity and that treatment of this underlying morbidity will resolve their FA-ship thereby reducing healthcare costs of FAs.21, 22 However, our model suggests that systematic detection and treatment of depression and anxiety after 1 or 2 years of frequent attendance results in only limited cost savings (2.5% at maximum) in comparison with the reference scenario. Baas et al showed that systematic screening for depression in a high risk population including FAs was not effective, because of low rates of treatment initiation.32 We assumed that uptake of treatment was 100%, but in clinical WHY DO THEY KEEP COMING BACK? practice this will probably be much lower. Therefore, the effects of systematic detection and treatment of depression and anxiety in frequent attenders were probably overestimated in this study. Assumptions in the model Firstly, we assumed that people who moved to a non-FA state remained in that state indefinitely. The most important reason for this choice was that we did not have enough data to estimate the transitions from a non-frequent attender state to a 1-year frequent attender state reliably. In addition, incorporation of this transition would have greatly complicated the model by further increasing the number of possible transitions. We expect that this assumption resulted in an overestimation of the potential benefits of systematic detection and treatment of psychiatric morbidity in FAs. Secondly, many frequent attenders stop being a frequent attender after 1 year, because the problem causing the frequent attendance is resolved within this first year. However, it may also be hypothesized that the longer the duration of FA-ship, the greater the chance that s/he will become a FA again. However, we did not have data available to include this effect. Therefore, we limited the time horizon of the model to 5 years. We expect that a longer time horizon would have led to less favourable results. Thirdly, we assumed that patients could have only one disorder at a time to avoid complicating the model further. However, presence of depression and anxiety simultaneously may negatively influence the success rate of treatment, thus leading to an overestimation of benefits in terms of costs and effects (Alpert, J; 2001). Strengths and weaknesses We modeled fixed risk reductions of treatment on psychiatric morbidity of 10%, 20% and 40%. This range is in line with recovery rates of psychotherapy reported in the literature.30 This can be considered strength of this study, because it indicates the boundaries of the effects that can be potentially gained by systematic detection and treatment of depression and anxiety in frequent attenders. Another important strength is that transition probabilities were estimated using data from a prospective cohort study among frequent attenders. Although some transitions were rare and could not be estimated precisely, we nevertheless expect that this study design feature greatly enhances the generalizability of our results. However, there are also a number of limitations to our study. First, only healthcare costs were included. However, absenteeism costs may be an important contributor to the total societal costs imposed by frequent attenders. Thus, our estimates may underestimate the potential cost savings, since one might expect that scenarios with larger health benefits also result in lower costs due to absenteeism. Second, diagnoses of depression and anxiety were based on self-report using the PHQ questionnaire and not on a clinical diagnostic instrument. A recent review, however, showed that the PHQ performs well as a screening instrument for depression in high risk groups such as FAs.33 The performance of the PHQ in diagnosing anxiety disorders, however, is only moderate.34 Thus, we probably missed some cases of depression and anxiety, while we also misclassified some CHAPTER 9 147 patients as being depressed or anxious. However, since the PHQ is also likely to be used in primary care when initiating systematic detection, we expect that our results are a good estimation of the potential effects of systematic detection and treatment of psychiatric morbidity in FAs. Finally, following recent Dutch primary care guidelines we assumed that treatment of psychotherapy consisted of short-lasting psychotherapy. Provision of psychotherapy is more expensive than prescription of antidepressants. However, the effect of antidepressants is probably overestimated in depression, although people with anxiety disorders probably respond better to antidepressant treatment. Thus, modelling of treatment with antidepressants would have resulted in lower treatment costs, but this probably has to be balanced against smaller effects. carefully weighed whether the potential benefits outweigh the efforts required from GPs to implement such a programme. Future research should indicate which treatments are effective in treating psychiatric morbidity in FAs, whether it is reasonable to assume spillover effects of such treatments on FA status and whether there are specific subgroups, such as e.g. FAs who express a need for treatment, in whom treatment is more effective. Conclusion and recommendations In conclusion, systematic diagnostic assessment and treatment of depression and anxiety in FAs results in minimal cost savings and health effects in comparison with FAs receiving usual care in a wellserviced multi-ethnic primary care setting. Moreover, uncertainty surrounding the cost and effect estimates was large, precluding strong conclusions. Based on the results of this study, systematic screening and treatment of psychiatric morbidity does not seem indicated. Such programmes can only be considered cost-effective or dominant in comparison with usual care if these treatments are associated with a spillover effect on FAship. However, even then it should be 148 WHY DO THEY KEEP COMING BACK? References 1. Neal, R.D., et al., Frequency of patients’ consulting in general practice and workload generated by frequent attenders: comparisons between practices. Br.J Gen.Pract, 1998. 48(426): p. 895-898. 2. Smits, F.T., et al., Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC. Public Health, 2009. 9: p. 36. 3. Little, P., et al., Psychosocial, lifestyle, and health status variables in predicting high attendance among adults. Br J Gen Pract, 2001. 51(473): p. 987-94. 4. Smits, F.T., et al., Defining frequent attendance in general practice. BMC.Fam Pract, 2008. 9: p. 21. 5. Vedsted, P. and M.B. Christensen, Frequent attenders in general practice care: a literature review with special reference to methodological considerations. Public Health, 2005. 119(2): p. 118-137. 6. Kersnik, J., I. Svab, and M. Vegnuti, Frequent attenders in general practice: quality of life, patient satisfaction, use of medical services and GP characteristics. Scand J Prim Health Care, 2001. 19(3): p. 174-7. 11. Vrca, B.M., et al., Frequent attenders in family practice in Croatia: retrospective study. Croat.Med J, 2004. 45(5): p. 620624. 12.Carney, T.A., S. Guy, and G. Jeffrey, Frequent attenders in general practice: a retrospective 20-year follow-up study. Br.J Gen.Pract, 2001. 51(468): p. 567-569. 13.Andersson, S.O., et al., Is frequent attendance a persistent characteristic of a patient? Repeat studies of attendance pattern at the family practitioner. Scand.J Prim.Health Care, 2004. 22(2): p. 91-94. 14.Koskela, T.H., O.P. Ryynanen, and E.J. Soini, Risk factors for persistent frequent use of the primary health care services among frequent attenders: a Bayesian approach. Scand J Prim Health Care, 2010. 28(1): p. 55-61. 15.Naessens, J.M., et al., Predicting persistently high primary care use. Ann Fam Med, 2005. 3(4): p. 324-30. 16.Rifel, J., et al., Association of common mental disorders and quality of life with the frequency of attendance in Slovenian family medicine practices: longitudinal study. PLoS One, 2013. 8(1): p. e54241. 17. Smits, F.T., et al., Interventions on frequent attenders in primary care. A systematic literature review. Scand.J Prim.Health Care, 2008. 26(2): p. 111-116. 7. Svab, I. and L. Zaletel-Kragelj, Frequent attenders in general practice: a study from Slovenia. Scand J Prim Health Care, 1993. 11(1): p. 38-43. 18.Adam, P., et al., Effects of team care of frequent attenders on patients and physicians. Fam Syst Health, 2010. 28(3): p. 247-57. 8. Smits, F.T., et al., Morbidity and doctor characteristics only partly explain the substantial healthcare expenditures of frequent attenders: a record linkage study between patient data and reimbursements data. BMC Fam Pract, 2013. 14: p. 138. 19.Bellon, J.A., et al., Successful GP intervention with frequent attenders in primary care: randomised controlled trial. Br.J.Gen.Pract., 2008. 58(550): p. 324330. 9. Ferrari, S., et al., Frequent attenders in primary care: impact of medical, psychiatric and psychosomatic diagnoses. Psychother Psychosom, 2008. 77(5): p. 306-14. 10.Ward, A.M., et al., Stability of attendance in general practice. Fam.Pract., 1994. 11(4): p. 431-437. 20.Barsky, A.J., et al., A Randomized Trial of Treatments for High-Utilizing Somatizing Patients. J Gen Intern Med, 2013. 21.Katon, W., et al., Distressed high utilizers of medical care. DSM-III-R diagnoses and treatment needs. Gen Hosp Psychiatry, 1990. 12(6): p. 355-62. CHAPTER 9 149 22.Dowrick, C.F., J.A. Bellon, and M.J. Gomez, GP frequent attendance in Liverpool and Granada: the impact of depressive symptoms. Br J Gen Pract, 2000. 50(454): p. 361-5. 23.Spitzer, R.L., K. Kroenke, and J.B. Williams, Validation and utility of a self-report version of PRIME-MD: the PHQ primary care study. Primary Care Evaluation of Mental Disorders. Patient Health Questionnaire. JAMA, 1999. 282(18): p. 1737-1744. 24.Smeets, H.M., N.J. de Wit, and A.W. Hoes, Routine health insurance data for scientific research: potential and limitations of the Agis Health Database. J Clin Epidemiol, 2011. 64(4): p. 424-430. 25.Baas, K.D., et al., Brief cognitive behavioral therapy compared to general practitioners care for depression in primary care: a randomized trial. Trials, 2010. 11: p. 96. 31.Gilchrist, G. and J. Gunn, Observational studies of depression in primary care: what do we know? BMC Fam Pract, 2007. 8: p. 28. 32.Baas, K.D., et al., Screening for depression in high-risk groups: prospective cohort study in general practice. Br J Psychiatry, 2009. 194(5): p. 399-403. 33.Wittkampf, K.A., et al., The psychometric properties of the panic disorder module of the Patient Health Questionnaire (PHQ-PD) in high-risk groups in primary care. J Affect Disord, 2011. 130(1-2): p. 260-7. 34.Wittkampf, K., et al., The accuracy of Patient Health Questionnaire-9 in detecting depression and measuring depression severity in high-risk groups in primary care. Gen.Hosp.Psychiatry, 2009. 31(5): p. 451-459. 26.Hakkaart-van Roijen, L., S.S. Tan, and C.A.M. Bouwmans, Handleiding voor kostenonderzoek: Methoden en standaard kostprijzen voor economische evaluaties in de gezondheidszorg. Geactualiseerde versie 2010. [Dutch manual for costing in economic evaluations]. 2011, College voor zorgverzekeringen (CVZ): Diemen. 27.Grol, R., et al., Attributes of clinical guidelines that influence use of guidelines in general practice: observational study. BMJ, 1998. 317(7162): p. 858-861. 28.Briggs, A.H., et al., Probabilistic analysis of cost-effectiveness models: choosing between treatment strategies for gastroesophageal reflux disease. Med Decis.Making, 2002. 22(4): p. 290-308. 29.Stinnett, A.A. and J. Mullahy, Net health benefits: a new framework for the analysis of uncertainty in costeffectiveness analysis. Med Decis Making, 1998. 18(2 Suppl): p. 68-80. 30.Cuijpers, P., et al., The effects of psychotherapies for major depression in adults on remission, recovery and improvement: A meta-analysis. J Affect Disord, 2014. 159C: p. 118-126. 150 WHY DO THEY KEEP COMING BACK? CHAPTER 9 151 general discussion 1. General discussion The aim of this thesis was to provide more insight in frequent attenders (FAs) and persistence of frequent attendance in Dutch General Practice. Our main objectives were to define a method to select FAs in a normal practice situation, to study morbidity of (persistent) FAs, to determine the workload and costs of healthcare use of (persistent) FAs, to examine whether it would be possible to predict persistence of frequent attending and to determine which in particular psychosocial factors may explain this persistence. Finally we reviewed the literature on effective treatments for FA and we evaluated whether detection and treatment of depression and anxiety after one or two years of frequent attendance might be cost-effective compared to usual GP care as provided in The Netherlands. 2. Results Part 1. Mapping frequent attenders in primary care The first research question addressed how to select FAs in a regular GP practice. FAs are patients who consult their general practitioner (GP) much more often than their peers. As consultation frequency is strongly dependent on sex and age, we used a proportional definition and defined FAs as the top 10% of attenders per sex and age group. Using such a definition also had the advantage of comparability with other countries, regions and practices. To calculate attendance we used all face-to-face contacts with the GP during one year (consultations in the office and house-calls) and the attendance rates of all enlisted patients. We showed that, in a real life GP practice situation, dividing the practice population into, at least, three age-bands per gender is an acceptable method to select FAs, which can be used by practicing GPs.1 152 WHY DO THEY KEEP COMING BACK? For the second research question we investigated the somatic, psychological and social problems of FAs. What are the differences between short-term and longterm FAs in this respect? What is the workload of a GP caused by (persisting) FAs? A retrospective study in the Hag-net-AMC database showed that most frequent attendance is temporary: Of all FAs during one year (1yFAs), 15.4 % persisted in frequent attendance during two consecutive years (persistent FA), which equaled 1.6% of all enlisted patients. However, the influence of FAs on the GP’s workload was substantial: Contacts of 1yFAs (by definition 10% of all enlisted patients) accounted for 39% of all face-to-face consultations; the contacts of the 1.6% of enlisted patients who became persistent FAs accounted for 8% of all consultations. Persistent FAs suffered more from social problems, feelings of anxiety, addictive behavior, and medically unexplained physical symptoms than 1yFAs and nonfrequently attending patients (non-FAs). They differed less from 1yFAs and non-FAs where the prevalence of chronic somatic diseases is concerned.2 The third research question addressed which readily available information noted in the patients’ Electronic Medical Record (EMD) predicts persistence of frequent attendance during 3 consecutive years (pFA). We showed that out of 3045 1yFAs, 470 (15.4%) became pFA. We selected indicators that were associated with becoming a pFA and constructed a prediction rule to predict which types of patients become pFA. With this prediction rule it was possible to change the prior probability of 15.4% to 3.3% (lowest value) or 43.3% (highest value), although the 10th and 90th centiles were 7.4 and 26.3%, respectively. The area under the receiver operating characteristics curve was 0.67 (c-statistic; 95% confidence limits 0.64 and 0.69). Using general cut-offs, our rule only moderately predicted which short-term FA continued to frequently attend in consecutive years.3 The fourth research question concerned a validation of this prediction rule in another time frame in the same GP database and in another GP database in a different part of the Netherlands. Our validation study in a different part of the Netherlands (Eindhoven; the SMILE database of general practice4) and in the same database but over a different timeframe (2009-2011), confirmed the results of the original study. The existing model (c-statistic 0.67) discriminated moderately with predicted values between 7.5 and 50%and c-statistics of 0.62 and 0.63, for validation in the original database and SMILE database, respectively. Data taken from the GP’s electronic medical records were only moderately indicative for which short-term FAs continued to frequently attend in consecutive years.5 The fifth and sixth research questions addressed the costs of health care of FAs in primary and specialist care. Can these costs be explained by the morbidities of these FAs and/or by GP characteristics? Unadjusted mean 3-year expenditures were 5044 and 15 824 Euros for non-FAs and pFAs, respectively. We showed that after adjustment for all included confounders, costs both in primary and specialist care remained substantially higher and increased with longer duration of frequent GENERAL DISCUSSION 153 attendance. As compared to non-FAs, adjusted mean 3-year expenditures were 1723 and 5293 Euros higher for 1yFA and pFAs, respectively. Thus, these extra costs could only partly be explained by the increased morbidity of (p)FAs as registered by the GP. Our results suggested only little influence of GP characteristics on costs of (persistent) frequent attenders. These increased costs might be explained by inadequate patient-GP communication and (as yet) undiagnosed psychosocial morbidity.6 Part 2. Review of the literature The seventh research question was to identify effective interventions to improve quality of life and lower attendance rate of FAs. This question was answered by a review of the literature to determine possible effective interventions to improve quality of life and lower attendance rate of FAs. Although we intended to perform a meta-analysis we were not able to pool the results because of the heterogeneity of the included studies. No study showed convincing evidence that an intervention improved quality of life or morbidity of frequent attending primary care patients, although a small effect might be possible in a subgroup of depressed frequent attenders. No evidence was found that it is possible to influence healthcare utilization of FAs.7 A intervention study, published after this review, which had a broad scope (a group of GPs assessing the reasons as to why a patient is frequently attending with subsequent targeted therapeutic measures) resulted in a significant and relevant reduction in frequent-attender consultations.8 Part 3. A prospective study of frequent attenders The eighth research question addressed which (in particular psychosocial) factors are associated with persistence of frequent attendance in a prospective cohort of incident1yFAs. Is there a supra-additive effect of combinations of somatic, psychological and social factors? Epidemiological studies of FAs showed that psychological problems (depression, anxiety, somatoform problems) are all more prevalent in FAs compared to non-FAs.2;9;10 However, in prospective cohort studies, using a proportional definition of FA (the upper 10%), only psychological distress, low physical quality of life and a low educational level predicted persistence of frequent attendance over the next two consecutive years.11;12 In our prospective cohort of incident (new) frequent attenders, we confirmed the association of persistence of frequent attendance with psychological distress. We showed that psychological determinants (panic, other anxiety, negative life events, illness behavior, and low mastery, but not depression) are associated with persistence of frequent attendance. We found no evidence of synergistic effects of somatic, psychological and social problems and no strong evidence of effects of GP characteristics on persistence of frequent attendance. The ninth research question evaluated whether systematic detection (as measured 154 WHY DO THEY KEEP COMING BACK? by the Patient Health Questionnaire) and treatment of depression and anxiety after one or two years of frequent attendance might be cost-effective compared to usual GP care. Based on a cost-effectiveness analysis with data of the prospective cohort of chapter eight using a Markov simulation we concluded that systematic detection and treatment of depressed and/or anxious one-year- and two-year-FAs are unlikely to be cost-effective compared with usual GP care as provided in the Netherlands. 3. In summary Patients with an age and sex-adjusted attendance rate ranking in the top 10 centile within a time frame of one year have more and multiple, somatic, psychological and social problems compared to their non-frequently attending peers. Somatic and psychosocial morbidity increases with longer duration of frequent attendance. Most frequent attendance is temporary and only about 2 % of the practice populations visits frequently during 3 or more consecutive years. Information from GPs’ electronic files is only a moderate indicator to identify which short-term FAs continue to frequently attend in consecutive years. Frequent attenders, and in particular persistent frequent attenders, make more use of primary care services than non-FAs. Frequent attenders, and in particular persistent frequent attenders, also have considerably above-average costs in primary and specialist healthcare in comparison with non-FAs. Morbidity of the patient and characteristics of the GP do not fully explain these extra costs. Panic complaints, general anxiety, life events, illness behavior and low mastery are associated with persistence of frequent attendance. These morbidities and characteristics are not (always) registered by the GP and may, partly, explain these extra costs. Also personality characteristics and patient-physician communication may play a role. An extrapolation of the results from our cohort study using a Markov simulation among 1yFAs showed that systematic detection and treatment of depression and/or anxiety is unlikely to be cost-effective in comparison with usual GP care. 4. Strengths of this thesis In this section we discuss the overall strengths of the work brought together in this thesis. For detailed information about a specific part of our research, please refer to the specific chapter. Despite many studies in other countries with a strong GP system, to our knowledge this is the first attempt at studying characteristics of FAs in the Netherlands. GENERAL DISCUSSION 155 Most research presented in this thesis was embedded in the longitudinal GP database of the department of General Practice, AMC (Hag-net-AMC). In this database, GPs register medical problems over a long period of time. This database of GP-patients, combined with a prospective cohort, identified through this database, and with a health insurers’ database (with detailed information about healthcare expenditures), made it possible to answer research questions 2 to 6 (part I) and 8 and 9 (part III). As all GPs participating in the Hag-net-AMC receive regular feedback on their registration activities and, nowadays, are helped by automated computer algorithms, these registration data are of reasonable quality, especially for somatic and psychological problems.13-15 It is less clear to what extent GPs register social problems adequately. Since GPs register problems during consultations, we expect the quality of this registration to be higher for FAs (who have many consultations). However, differences in prevalence figures of diseases between registration networks are considerable and limit generalisation.15 These differences may be caused by differences between regions, or by registration and coding differences between GPs or by artefacts.15;16 Being a long-time member of the steering committee of HAG-net-AMC and personally knowing many of the participating GPs, helped to secure participation and to optimise data quality. When convincing practices to participate in the (continuation of) our study or to complete missing data, it was beneficial to have an experienced, local GP to lead the study. 5. Limitations of this thesis In this section we discuss the general limitations of the work laid down in this thesis. For detailed descriptions, please refer to the specific chapter. First, we used a proportional definition of FA which has certain disadvantages: some patients may drop just below the cut-off (89th centile e.g.) the next year. However, definitions based on some absolute number of visits share this limitation (chapter 2). Second, we assumed that the so-called Problem List (a list of all patients’ problems as registered and coded by the GP in the electronic medical record) would give a comprehensive picture of the main morbidity of a patient. However, some registered problems can be overreported if resolved problems are not removed from this list or underreported if a prevalent problem is not registered on the Problem List by the GP. This could lead to over- and underestimation of the prevalence of these disorders and to information bias in the estimations of the effect on persistence of frequent attendance.17 Also GP differences in registration- and coding-discipline may be confounding factors.13;14 It is also unclear whether and to what extent FAs visit primary care for minor, transient, problems not captured by the Problem List. 156 WHY DO THEY KEEP COMING BACK? Third, the participating GPs appeared to have relatively similar characteristics: Being embedded in one academic region, eighty-two percent of the GPs were involved in educating medical students, vocational training of future GPs and/or research activities. This may lead to non-detection or underestimation of the effects of GP characteristics. Fourth, although we attempted to collect complete sets of patient characteristics and clinical data, other undocumented determinants of (p)FA may have been present. This is particularly true for the retrospective database studies, (part I) in which we had no influence on what was documented. Fifth, this study is situated in an urban part of the specific Dutch healthcare system with more than average Surinamese and Ghanese patients. This may limit the extent to which our results can be generalised to other regions and countries. Sixth, in the prospective cohort study (part III), about two thirds of the eligible 1yFAs refused to participate. However, because we focused on aetiology and ample analytical contrast within our sample, representativeness was not an issue like, for instance, in a survey predicting some election and we do not think this influenced our results negatively.17 Nonetheless, it was surprising how sensitive these patients appear to being called a FA. From our personal contacts with participating FAs we got the impression that they did not recognise themselves in the label ‘FA’. The term may be seen as pejorative and blaming to the patient.18-20 Therefore, in a British study a user panel preferred the term “regular attender”.21 Finally, we analysed a cohort of new (incident) FAs, and our results are limited to frequent attendance over a period of 3 years. We cannot comment on frequent attending of longer durations. 6. Relevant literature Frequent attendance has interested GPs, mostly in countries which use (largely) a capitation system for remuneration and where GPs use a list system. Over the last thirty years, a continuous flow of mostly descriptive articles about FAs has been published from the United Kingdom, Scandinavian countries, Spain and from Health Maintenance Organisations in the United States. Surprisingly, despite having largely a capitation payment system the Netherlands has been an exception. The only Dutch study we know of is a thesis about the association between personality and medical consumption (1980).22 That study indicated that “personality” (defined as neuroticism, somatisation) only increased medical consumption in the top 5% of attenders. As expected, results in the literature differ depending on the definition of frequent attendance used. When no correction for age and sex is used the resulting GENERAL DISCUSSION 157 overrepresentation of elderly and female patients results in a high prevalence of somatic (multi)morbidity. Using the proportional definition on the other hand, results in a better representation of all ages and both sexes resulting in higher prevalence of psychological and social problems than without correction for age and sex. In several studies, FAs are used to select a group of patients at high risk for distress23, mental health problems24, major depressive disorder25-27 or somatoform disorders/ medically unexplained symptoms28;29, but not for anxiety. Generally, screening- and treatment programs using frequent attendance as a selection criterion to diagnose a target disease had disappointing results in improvement of this target disease, 7;25;30 partly because uptake and acceptance of screening and treatment in screened patients are generally low.31-33 In our opinion, using FAs to select a certain disease or problem disregards the strong multi-causal character of frequent attendance. Detection and treatment of just one underlying aspect is not likely to resolve the complex, multiple problems found in FAs. In this context, it is noteworthy that there is a wide range of studies about comorbid depression in somatic diseases, but that such knowledge is much less for anxiety as a comorbidity. Reviewing the literature we found no evidence that specific interventions reduce healthcare utilization or improve quality of life of FAs in comparison with usual GP care.7 However, one more recent Spanish intervention study of good quality in 1yFAs showed that a GP intervention reduced attendance in primary care in comparison with usual care. Bellón et al. applied a “7 Hypothesis + Team intervention” after 3 GPs received a 15h training in executing the intervention. The GPs held meetings to share analyses and reflections on their FAs and to make tailored plans for each FA. GPs also received emotional support in these meetings and were helped to generate strategies to deal with FAs. Numbers of consultations decreased by 5.08/ year (p<.001).8 7. Clinical and public health implications This thesis distinguishes between short-term and persistent frequent attendance. Our results imply that in short-term FAs, persistence of frequent attendance is associated with anxiety, illness behavior, negative life events and low mastery. Thus, a proportion of pFA may be preventable by developing interventions to diminish or eliminate these causes.34-39 Adequate detection and treatment of these conditions in FAs may result in a better quality of life of these patients, less morbidity and a decrease in attendance and costs. Although treatment of anxiety in primary care patients has been shown to be effective, the effect of this treatment in preventing persistence of FAs is unclear and needs to be demonstrated in research.7;34;40 In a first attempt to explore this issue, using a Markov model 158 WHY DO THEY KEEP COMING BACK? extrapolating the results of the prospective cohort study, we showed that systematic diagnostic assessment and treatment of depression and anxiety in FAs will probably result in minimal cost savings and health effects in comparison with FAs receiving usual care in primary care (chapter 9). However, knowing the high costs of (persistent) FAs it might be beneficial for these patients and for the budget problems in healthcare to test other interventions in (p)FAs. 7 GP interventions to better the (medical) situation of FAs should target these patients broadly, at least including interventions which address present panic and anxiety complaints and low mastery. In addition, knowing the results of Bellon, interventions to diminish consultations frequency of FAs may also need to be targeted at the GP .8 The Frequent attender: a typical GP patient Currently, most research in general practice is focussed on individual, well defined, mono-causal issues and diseases. From a methodological perspective this is understandable. Diabetes, asthma or depression are easier research topics than multi-morbidity, headaches, deprivation and ……frequent attendance. Similarly, in clinical general practice, GPs often prefer to focus on clear diseases, despite the sometimes low prevalence and/or lack of therapeutic options (e.g. in the case of COPD). The traditional diagnostic, treating, and consoling role of the life-long physician is more and more superseded by large-scale preventive and intervention programs. Large amounts of funding and effort are spent on programs reaching a relatively small number of patients, sometimes without much scientific evidence. We think these trends disregard the complicated reality of everyday general practice. The 10% most frequently attending patients consume 40% of the GP’s consultation time. These FAs are an important and, as shown in our study, expensive group of patients in general practice. Often, these FAs have complicated problems and suffer from a mix of interacting social, psychological and medical problems, which often cannot be classified in somatic or psychiatric (DSM-IV) diagnoses. Only a GP with a broad bio-psychosocial knowledge and approach is able to unravel this mix of problems and propose suitable treatment and guidance. Research has shown that even then, the proposed treatment has to suit the perception, expectations and wishes of the patient to be effective.32;41 In our opinion, such an integral approach is a better match for FAs than the preprogrammed approach of disease programs. Since in countries with a strong primary care orientation, the system’s focus is on continuity of care, knowledge about the (family) background of the patient and bio-psychosocial care, the GP can best be the backbone of such an integral approach of FAs, if necessary supported by a psychologist. In this regard, the new approach in mental health care, where a formal DSM- diagnosis is needed before treatment by a psychologist GENERAL DISCUSSION 159 or psychotherapist can start, can be seen as a, possibly counterproductive and costly downfall.36 The results of our prospective cohort study also urge for more emphasis on anxiety problems in everyday general practice. The role of lack of mastery in persistence of frequent attendance calls for experiments to try to strengthen coping mechanisms of FAs. Possibly social interventions in FAs (targeted at assertiveness, housing and debts problems) may have better results than medical interventions. Stepped care approach We support the approach of a multifaceted diagnostic process and treatment for frequent attending patients, as advocated by Bellon.8 Unlike his approach, we think such a program may be preferably directed at frequent attenders of longer duration (e.g. 2yFAs). Such a program must be tested first in a proper randomized clinical trial. Until such results are available, we propose that General Practitioners identify their FAs, give them some sort of ‘label ‘in the electronic medical record (“ruiter”), and try to implement a broad diagnostic and therapeutic approach in their usual care of these patients. A diagnostic interview, possibly supported by a psychiatric questionnaire (PHQ, 4-DKL, HADS), can help the GP to unravel the somatic, psychological and social background of the patient, and to formulate a targeted therapeutic approach. 8. Implications for research We think further research should focus on the background of repeated or persistent frequent attendance. Also the role of the Problem List as proxy for FA’s morbidity needs more clarification. A cohort with more incident FAs, use of standardized psychiatric interviews and measures of quality of life may provide more insight in the difference between PHQ diagnoses and diagnoses after a Structured Clinical Interview for DSM Disorders (SCID diagnosis) and the role of Quality of life measures. Subsequently, randomised trials may determine the extent to which interventions aimed at modifying anxiety disorders, illness behavior and mastery in this specific group of patients are acceptable to patients and improve quality of life, and reduce attendance and costs of (persistent) FAs. Using a more diverse group of GPs (setting of the practice, age distribution, (non) training practices, (non) academic practices) could shed more light on the influence of the GP on (persistence of) frequent attendance. Let’s face the frequent attender not as heart sink but as a challenge for GP care! Frequent attenders deserve better care, not more care! 160 WHY DO THEY KEEP COMING BACK? References (1) Smits FT, Mohrs J, Beem E, Bindels PJ, van Weert HC. Defining frequent attendance in general practice. BMC Fam Pract 2008; 9(1):21. (2) Smits FT, Brouwer HJ, ter Riet G., van Weert HC. Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC Public Health 2009; 9(1):36. (3) Smits FTM, Brouwer H.J., ter Riet G, van Weert HC. Predictability of persistent frequent attendance. A historic 3-year cohort study. Br J Gen Pract 2009; 2-2009(59):114-119. (4) van den Akker M, Spigt MG, De Raeve L., van Steenkiste B., Metsemakers JF, van Voorst EJ et al. The SMILE study: a study of medical information and lifestyles in Eindhoven, the rationale and contents of a large prospective dynamic cohort study. BMC Public Health 2008; 2008:19. (5) Smits FT, Brouwer HJ, Zwinderman AH, van den Akker M, van SB, Mohrs J et al. Predictability of persistent frequent attendance in primary care: a temporal and geographical validation study. PLoS One 2013(9):e73125. (6) Smits FT, Brouwer HJ, Zwinderman AH, Mohrs J, Smeets HM, Bosmans JE et al. Morbidity and doctor characteristics only partly explain the substantial healthcare expenditures of frequent attenders: a record linkage study between patient data and reimbursements data. BMC Fam Pract 2013; 2013(1):138. (7) Smits FT, Wittkampf KA, Schene AH, Bindels PJ, van Weert HC. Interventions on frequent attenders in primary care. A systematic literature review. Scand J Prim Health Care 2008; 26(2):111-116. (8) Bellon JA, Rodriguez-Bayon A, de Dios LJ, Torres-Gonzalez F. Successful GP intervention with frequent attenders in primary care: randomised controlled trial. Br J Gen Pract 2008; 58(550):324-330. (9) Ferrari S, Galeazzi GM, Mackinnon A, Rigatelli M. Frequent attenders in primary care: impact of medical, psychiatric and psychosomatic diagnoses. Psychother Psychosom 2008; 77(5):306-314. (10) Gili M, Luciano JV, Serrano MJ, Jimenez R, Bauza N, Roca M. Mental disorders among frequent attenders in primary care: a comparison with routine attenders. J Nerv Ment Dis 2011(10):744749. (11) Rifel J, Svab I, Selic P, Rotar PD, Nazareth I, Car J. Association of Common Mental Disorders and Quality of Life with the Frequency of Attendance in Slovenian Family Medicine Practices: Longitudinal Study. PLoS One 2013; 8(1):e54241. (12) Vedsted P, Fink P, Olesen F, MunkJorgensen P. Psychological distress as a predictor of frequent attendance in family practice: A cohort study. Psychosomatics: Journal of Consultation Liaison Psychiatry /9; 42(5):416-422. (13) Brouwer HJ, Bindels PJ, van Weert HC. Data quality improvement in general practice. Fam Pract 2006; 23(5):529536. (14) van den Dungen C, Hoeymans N, Boshuizen HC, van den Akker M, Biermans MC, van BK et al. The influence of population characteristics on variation in general practice based morbidity estimations. BMC Public Health 2011; 11:887. (15) van den Dungen CHN, Boshuizen HC, van den Akker M, Biermans MC, van Boven K., Brouwer HJ et al. What factors explain the differences in morbidity estimations among general practice registration networks in the Netherlands? A first analysis. Eur J Gen Pract2008;14 Suppl 1:53-62 (16) Wennberg JE. Dealing with medical practice variations: a proposal for action. Health Aff (Millwood ) 1984; 3(2):6-32. (17) Rothman K, Greenland S, Lash T. Modern epidemiology. Philadelphia, PA (USA): Lippincott Williams & Wilkins; 2008. GENERAL DISCUSSION 161 (18) Neal RD, Heywood PL, Morley S. ‘I always seem to be there’--a qualitative study of frequent attenders. Br J Gen Pract 2000(458):716-723. (19) Hodgson P, Smith P, Brown T, Dowrick C. Stories from frequent attenders: a qualitative study in primary care. Ann Fam Med 2005; 3(4):318-323. (20) Dwamena FC, Lyles JS, Frankel RM, Smith RC. In their own words: qualitative study of high-utilising primary care patients with medically unexplained symptoms. BMC Fam Pract 2009; 10(1):67. (21) Morriss R, Kai J, Atha C, Avery A, Bayes S. Persistent frequent attenders in primary care: costs, reasons for attendance, organisation of care and potential for cognitive behavioural therapeutic intervention. BMC Fam Pract 2012; 13:39. (22) van der Ploeg HM. Persoonlijkheid en medische consumptie: Een onderzoek naar de relatie van persoonlijkheidsfactoren en de frequentie van huisartsbezoek. Swets &Zeitlinger B.V.; 1980. (23) Katon W, Von KM, Lin E, Bush T, Russo J, Lipscomb P et al. A randomized trial of psychiatric consultation with distressed high utilizers. Gen Hosp Psychiatry 1992; 14(2):86-98. (24) Schreuders B, van Marwijk H, Smit J, Rijmen F, Stalman W, van Oppen P. Primary care patients with mental health problems: outcome of a randomised clinical trial. Br J Gen Pract 2007; 57(544):886-891. (25) Katzelnick DJ, Simon GE, Pearson SD, Manning WG, Helstad CP, Henk HJ et al. Randomized trial of a depression management program in high utilizers of medical care. Arch Fam Med 2000; 9(4):345-351. (26) Baas KD, Wittkampf KA, van Weert HC, Lucassen P, Huyser J, van den Hoogen H et al. Screening for depression in high-risk groups: prospective cohort study in general practice. The British Journal of Psychiatry 2009; 194(5):399403. 162 (27) Berghofer A, Roll S, Bauer M, Willich SN, Pfennig A. Screening for Depression and High Utilization of Health Care Resources Among Patients in Primary Care. Community Ment Health J 2014:Jan. (28) Barsky AJ, Ahern DK, Bauer MR, Nolido N, Orav EJ. A Randomized Trial of Treatments for High-Utilizing Somatizing Patients. J Gen Intern Med 2013:Mar. (29) Smith RC, Gardiner JC, Armatti S, Johnson M, Lyles JS, Given CW et al. Screening for high utilizing somatizing patients using a prediction rule derived from the management information system of an HMO: a preliminary study. Med Care 2001; 39(9):968-978. (30) Schreuders B, van Oppen P, van Marwijk HW, Smit JH, Stalman WA. Frequent attenders in general practice: problem solving treatment provided by nurses. BMC (31) Vink JM, Willemsen G, Stubbe JH, Middeldorp CM, Ligthart RS, Baas KD et al. Screening for depression in high-risk groups: prospective cohort study in general practice. Eur J Epidemiol 2004; 2004;19(7):623-630. (32) Wittkampf KA, van Zwieten M, Smits FT, Schene AH, Huyser J, van Weert HC. Patients’ view on screening for depression in general practice. Fam Pract 2008; 25(6):438-444. (33) Thombs BD, Ziegelstein RC, Roseman M, Kloda LA, Ioannidis JP. There are no randomized controlled trials that support the United States Preventive Services Task Force guideline on screening for depression in primary care: a systematic review. BMC Med 2014:13-12. (34) Kendall T, Cape J, Chan M, Taylor C. Management of generalised anxiety disorder in adults: summary of NICE guidance. BMJ 2011; 2011(4):318-327. (35) Pilling S, Mayo-Wilson E. Recognition, assessment and treatment of social anxiety disorder: summary of NICE guidance. BMJ 2013; 346. (36) Feltz-Cornelis CM, Van Oppen P, Ader H, Van Dyck R. Randomised controlled trial of a collaborative care model with psychiatric consultation for persistent medically unexplained symptoms in general practice. Psychother Psychosom 2006; 75:282-289. WHY DO THEY KEEP COMING BACK? (37) van der Feltz-Cornelis C, Van Os TWDP, Van Marwijk HWJ, Leentjens AFG. Effect of psychiatric consultation models in primary care. A systematic review and meta-analysis of randomized clinical trials. Journal of Psychosomatic Research 68[6], 521-533. 2010. (38) Reger MA, Gahm GA. A metaanalysis of the effects of Internet- and computer-based cognitive-behavioral treatments for anxiety. Journal of Clinical Psychology 2009; 65(1):53-75. (39) Donker T, Griffiths KM, Cuijpers P, Christensen H. Psychoeducation for depression, anxiety and psychological distress: a meta-analysis. BMC Med 2009; 7:79.:79. (40) Goldberg D. The “NICE Guideline” on the treatment of depression. Epidemiologia e Psichiatria Sociale Vol 15 (1), Jan -Mar 2006;-15. (41) Arroll B, Goodyear-Smith F, Kerse N, Fishman T, Gunn J. Effect of the addition of a “help” question to two screening questions on specificity for diagnosis of depression in general practice: diagnostic validity study. BMJ 2005(7521):884.Fam Pract 2005; 6:42. GENERAL DISCUSSION 163 chapter 11 appendices Appendix 1. Selected problems and diseases with ICPC-code1 ICPC code Problem Diabetes T90 Diabetes mellitus type 1 and 2 Chronic Cardiovascular disease K74 Angina pectoris K75 Acute myocardial infarction K76 Other and chronic ischemic heart disease K77 Heart failure K78 Atrial fibrillation/flutter K82 Pulmonary heart disease K83 Heart valve disease, non-rheumatic2 K86 Hypertension, uncomplicated K87 Hypertension with involvement of target organs K89 Transient cerebral ischemia K90 Cerebrovascular accident; stroke K91 Atherosclerosis excluding heart and brain K92 Other arterial obstructive/ peripheral vascular disease Chronic respiratory problems R70 Tuberculosis of respiratory organs R91 Chronic bronchitis/ bronchiectasis R95 Emphysema/Chronic Obstructive Pulmonary Disease R96 Asthma R97 Allergic rhinitis; hay fever 164 WHY DO THEY KEEP COMING BACK? Psychological / Psychiatric problems All P All P-codes Sub-category 1: Anxious feelings P01 Feeling anxious/nervous/tense P74 Anxiety disorder/anxiety state P09 Concern sexual preference A-Y27 Fear of other disease of various tracts A-D26 Fear of cancer of various tracts L26 Fear of death N26 Fear of AIDS R-Y26 Fear of heart attack A25 Fear of hypertension B25 Fear of venereal disease K24 Fear of sexual dysfunction K25 Fear of genital cancer X23/Y25 Fear of venereal disease X24/Y24 Fear of sexual dysfunction X25 Fear of genital cancer Sub-category 2: Depressed feelings P03 Feeling depressed P76 Depressive disorder Sub-category 3: Addictive behaviour P15 Chronic alcohol abuse P16 Acute alcohol abuse P17 Tobacco abuse P18 Abuse of medicines P19 Drug abuse Continued on page 166 APPENDICES 165 Social problems All Z All socio-economic problems Medically unexplained physical problems (MUPS) L01 Neck symptoms /complaints (excluding headache) L02 Back symptoms/complaints L03 Low back complaints without radiation L04 Chest symptoms/complaints L18 Muscle pain/ fibromyalgia N01 Headache N02 Tension headache A04 General weakness/tiredness/chronic fatigue P06 Disturbance of sleep/insomnia P20 Disturbance of memory, concentration T03 Loss of appetite T07 Weight gain T08 Weight loss P04 Feeling/behaving irritably R02 Shortness of breath, dyspnea K04 Palpitations/ aware of heartbeat N17 Vertigo/Dizziness R21 Symptoms / complaints of the throat N06 Other sensitive disturbance /abnormal involuntary movements D09 Nausea D11 Diarrhea / Loose bowels D08 Flatulence/gas pain/belching D12 Constipation D01 Generalized abdominal pain/cramps D93 Irritable bowel syndrome 166 WHY DO THEY KEEP COMING BACK? Cancer A79 Cancer of unknown location B72-B74 Cancer of hematological tract D74-D77 Cancer of stomach, colon, pancreas K72 Cancer of the vascular tract L71 Cancer of the locomotor tract N74 Cancer of the nervous system S77 Cancer of the skin T71,T73 Cancer of the thyroid, endocrine tract U75-U77 Cancer of the kidney, bladder, urine tract W72 Cancer related to pregnancy X75-X77 Cancer of the female organs Y77-Y78 Cancer of the male organs All D All digestive tract problems/diseases All L All locomotor tract problems/diseases All S All skin problems/diseases 1: 2: International classification of primary care Not otherwise specified Appendix 2. Effect of loss to follow-up and clustering on the health centre level on the prognostic index. Amsterdam I Amsterdam II SMILE Deviance of the normal Logistic Regression model 2476 3046 5320 Ln(OR)1 of the prognostic index (SE) 0.993 (0.084) 0.663 (0.071) 0.835 (0.062) Ln(OR) of the prognostic index corrected for Loss to follow-up (SE) 1.028 (0.080) 0.694 (0.067) 0.848 (0.062) Deviance of the multilevel model 2467 3029 5198 Ln(OR) of the prognostic index multilevel model (SE) 0.994 (0.085) 0.707 (0.073) 0.909 (0.064) variance practice effect 0.072 0.102 0.222 Intraclass correlation coëfficiënt 0.0214 0.0301 0.0632 1 2 2 Indicates natural logarithm of the odds ratio SE indicates Standard Error APPENDICES 167 Appendix 3. Primary care physicians’ questionnaire The interaction between patient and physician factors may determine the number of visits of a patient. With this questionnaire we are trying to determine primary care physicians’ characteristics which may influence patients’ attendance. Filling out this survey will take about 10 minutes of your time. Please answer all questions. After entering, your data will, of course, be anonymized. Thank you very much for your effort! 1. In which year did you graduate from medical school? 2. At what university did you graduate as a doctor? 3. Please note the number of years you work as a physician: … 4. I am currently working • part-time • full-time 5. If part-time, what percentage of a full contract do you work?: … 6. Do you have special knowledge of/ interest in a specific part of general practice? • Yes • No If so, in which specific field are you interested? (Several options are possible) • paediatric medicine • geriatric medicine • small surgical procedures • management of the practice • financial management • professional organizations 7. Do you have special knowledge of/ interest in (the treatment of) diabetes mellitus? • no special interest • little interest • normal interest • more than normal interest • very much interest 8. Do you have special knowledge of/ interest in (the treatment of) asthma and/ or COPD? • no special interest • little interest • normal interest • more than normal interest • very much interest 168 9. Do you have special knowledge of/ interest in (the treatment of) cardiovascular diseases? • no special interest • little interest • normal interest • more than normal interest • very much interest 10. Are you a member of a guideline committee of the Dutch College of General Practitioners (NHG)? • Yes • No 11. If “Yes”: Please give the title of the guideline(s) • NHG-standard: • NHG-standard: • NHG-standard: 12. Are you involved in the organization of continuous medical education/post academic training of primary care physicians? • Yes • No 13. Do you treat patients with anxiety disorders? Please indicate what percentage of patients with anxiety disorders you manage yourself. • 0% • 25% • 50% • 75% • 100% 14. Do you treat patients with depressive symptoms or depression? Please indicate what percentage of patients with depressive symptoms you manage yourself. • 0% • 25% • 50% • 75% • 100% WHY DO THEY KEEP COMING BACK? 15. Do you treat patients with medically unexplained somatic complaints? (Think for instance of unexplained pain) Please indicate what percentage of patients with unexplained symptoms you manage yourself. • 0% • 25% • 50% • 75% • 100% 16. Are you a primary care physician vocational trainer? • Yes • No 17. Do you regularly give (vocational) training to medical students ? • Yes • No 23. How do you organize your office schedule? Check on the checkboxes (multiple options). • Only after appointment • An open schedule • Telephone consultation • E-mail consultation • Appointments via the Internet 24. My age is: 25. I am: • Man • Woman Thank you very much for completing this questionnaire! 18. Are you also working at a university and/or the Dutch College of General Practitioners (NHG)? Multiple options are possible. • Yes, university • Yes, NHG • No 19. Do you cooperate with other primary care physicians (PCPs)? • Group practice (only PCPs) • Health centre • PCPs with one electronic medical file • I am a solo PCP 20. Do you work with a trained practice nurse? • Yes • No 21. If so, how many days support these practice nurses your practice? _ _ days 22. Do you work with another discipline? Please check the appropriate checkbox (multiple options). • Nurse • Social psychiatric nurse • General social worker • Primary care psychologist • Other: ….. APPENDICES 169 Appendix 4. Univariate associations between patient characteristics and 3-year costs in primary and specialist care Primary care Age1 Specialists care Mean (range) Difference (SE) P-value Difference (SE) P-value 47 (18-98) 59 (2) < 0.001 101 (6) < 0.001 Male (reference) 0 Female 343 (74) 0 < 0.001 155 (231) 0.50 Ethnicity: Dutch (reference) 0 0 Moroccan -409 (186) Turkish -369 (244) 0.001 -709 (764) 0.17 Surinamese -318 (96) 0.001 -190 (300) 0.17 < 0.001 1466 (50) < 0.001 < 0.001 0.001 -1212 (590) 0.17 Problems on the problem list2 Number of problems 2.43 (0-18) 743 (15) Diabetes 0.14 (0-1) 3146 (116) < 0.001 4003 (369) COPD/Asthma 0.16 (0-2) 1132 (96) < 0.001 929 (302) 0.002 Cardiovascular 0.42 (0-5) 1684 (52) < 0.001 4210 (165) < 0.001 Social 0.04 (0-2) 244 (212) 0.25 660 (649) 0.32 Psychological 0.24 (0-3) 1183 (81) < 0.001 4506 (254) < 0.001 Depression 0.06 (0-1) 1292 (179) < 0.001 2602 (559) < 0.001 Anxiety 0.03 (0-1) 886 (231) < 0.001 1423 (723) 0.049 Addiction 0.05 (0-2) 1235 (186) < 0.001 5617 (582) < 0.001 Other psychological 0.10 (0-2) 1404 (128) < 0.001 6711 (400) < 0.001 Medically Unexplained Symptoms 0.17 (0-5) 947 (96) < 0.001 1791 (300) < 0.001 Frequent attender during: Non-FAs (reference) 0 1 year 1005 (109) <0.001 2467 (355) <0.001 2 years 2227 (246) <0.001 3512 (461) 3 years 3029 (308) <0.001 7751 (1024) <0.001 1: 2: 170 0 <0.001 Costs per additional year of age Binary variables were modeled using a dummy; all other variables were modelled linearly WHY DO THEY KEEP COMING BACK? Appendix 5. The multivariate effects of patient characteristics on mean costs (by healthcare level#) Age1 Primary care Specialists care Mean (range) 4 difference (SE) P-value difference (SE) P-value 47(18-98) 8.1 (1.1) < 0.001 -4.5 (3.0) 0.13 Male (reference) 0 Female 119 (33) 0 < 0.001 259 (86) 0.002 Ethnicity Dutch (reference) 0 Moroccan 171 (131) 0 0.087 7 (343) Turkish -41 (11) 194 (290) Surinamese -36 (106) 38 (277) 0.54 Problems on the problem list ¶ Total Number 2.43 (0-18) 451 (17) < 0.001 802 (44) < 0.001 Diabetes mellitus 0.14 (0-1) 1330 (59) < 0.001 -268 (155) 0.083 COPD2/Asthma 0.16 (0-2) 161 (46) 0.001 -660 (121) < 0.001 Cardiovascular 0.42 (0-5) 263 (36) < 0.001 659 (94) < 0.001 Social 0.04 (0-2) -512 (95) < 0.001 -582 (249) 0.019 Psychological 0.24 (0-3) 365 (282) 0.196 1074 (740) 0.15 Depression 0.06 (0-1) -401 (292) 0.17 -433 (767) 0.57 Anxiety 0.03 (0-1) -472 (263) 0.072 -588 (689) 0.39 Addiction 0.05 (0-2) -344 (295) 0.24 -329 (775) 0.67 Other psychological 0.10 (0-2) 191 (289) 0.51 428 (758) 0.57 Medically Unexplained Symptoms 0.17 (0-5) -279 (50) < 0.001 -801 (131) < 0.001 Cancer 0.04 (0-1) 635 (82) < 0.001 4195 (217) < 0.001 Locomotor 0.16 (0-1) -81 (57) 0.15 290 (148) 0.05 Skin 0.09 (0-1) -289 (61) < 0.001 -1124(159) < 0.001 Digestive 0.11 (0-1) 113 (60) 0.059 492 (156) 0.002 Intraclass correlation (PCP level 3) #: ¶: 1: 2: 3: 4: 0.0097 0.0041 Based on the same regression analysis as presented in Table 4 All variables were linear, unless indicated otherwise Costs in Euros per unit increase (for instance per extra year of age) Chronic obstructive pulmonary disease Variance between primary care physicians (PCPs) as part of the total variance (residual variance + PCP variance). Frequent attenders during 1 year in 2009 . APPENDICES 171 Appendix 6. instruments used to capture the potential psychosocial aetiological factors for persistence of frequent attendance. 1. Do you worry about your health? 2. Are you worried that you may get a serious illness in the future? 3. Does the thought of a serious illness scare you? 4. If you have pain, do you worry you might have a serious illness? 5. If a pain lasts for a week or more do you see a physician? 6. If a pain lasts a week or more, do you believe you have a serious illness? 7. Do you avoid habits which may be harmful to you, such as smoking? 8. Do you avoid foods which may not be healthy? 9. Do you examine your body to see if there is something wrong? 10. Do you think that you have a illness but doctors have not diagnosed it? 11. When your doctor says you have no illness, do you refuse to believe it? 12. When your doctor tells you what was found, do you believe you have it? 13. Are you afraid of news which makes you think of death? 14. Does the thought of death scare you? 15. Are you afraid that you may die soon? 16. Are you afraid that you may have cancer? 17. Are you afraid that you may have heart disease? 18. Are you afraid that you may have another serious illness? 19. When you read or hear about an illness, do you get similar symptoms? 172 WHY DO THEY KEEP COMING BACK? Most of the time Often Sometimes Seldom No Illness attitude scale: Don’t think too long before answering the next questions: Just ask yourself does it happen and if it happens how often? 20. When you notice a sensation in your body, do you find it difficult to think of something different? 21. When you feel something in your body, do you worry about it? 23. Has your physician told you that you are currently suffering from a disease? Yes No *24. How often do you see a doctor? Almost never Seldom About 4 times a year About once a month About once a week *25. How many different caregivers (doctors, homoeopaths etc) have you seen in the past year? None 1 2 or 3 4 or 5 6 and more *26. How often have you been treated in the past year? Not at all Once 2 or 3 times 4 or 5 times 6 times or more 27. In the next three questions we ask you about your physical symptoms (for instance pain, tensions in your body, breathing problem, tiredness) 1. Do your bodily symptoms stop you from working? 2. Do your bodily symptoms stop you from concentrating? 3. Do your bodily symptoms stop you from enjoying yourself? * Question which counts for the health anxiety score Question which score for the illness behaviour score Questions which score for the illness behaviour score, omitted from the IAS to avoid circular reasoning APPENDICES 173 Stronglt disagree disagree Don’t agree/ don’t disagree Agree Strongly agree Mastery questionnaire How strongly do you agree or disagree with the following statements? 1. I have little control over the things that happen to me. 2. There is really no way I can solve some of the problems I have. 3. There is little I can do to change many of the important things in my life. 4. I often feel helpless in dealing with the problems of life. 5. Sometimes I feel that I’m being pushed around in life. 6. What happens to me in the future mostly depends on me. 7. I can do just about anything I really set my mind to do. Negative life events If you have never or not in the last 12 months experienced a certain event, please fill in the square below “Not applicable”. If you did experienced a certain event last year, please fill in the square below the time at which the event took place. If an event took place over several time periods, please fill in this event for all these time periods. 1. Divorce: a. close family member b. yourself c. significant other/s 2. New relationship of yourself 3. Moving house (yourself) 174 WHY DO THEY KEEP COMING BACK? Yes, 3 to 12 months ago Past year’s events: If Yes, when? (More than one answer possible) Yes, during the last 3 months 1. Close family members can be: parents, foster parents, brothers, sisters and children. 2. Significant others can be: friends, other family members, a trusted person, pastor, vicar or other people that are close to you Not applicable Please note: 4. Long-term and/or severe physical illness: a. close family member or partner b. yourself c. significant other/s 5. Death of: a. a close family member or partner f. significant other/s 6. Severe mental illness: a. close family member or partner b. yourself c. significant other/s 7. Attempted suicide: a. close family member or partner b. yourself c. significant other/s 8. Violence within your family or relationship 9. Alcohol or drug abuse within your family or relationship 10. Been a victim of a crime 11 Been a victim of a serious accident 12. Been a victim of sexual abuse 13. Been a victim of physical abuse 14. Had an unwanted pregnancy or have made someone pregnant who did not want to be 15. Problems concerning your work: a. conflicts/arguments b. dismissal 16. Financial troubles of yourself a. worries about managing your finances b. debts APPENDICES 175 Patient Health Questionnaire Nearly every day More than half the days Several days Not at all Depression: Over the last 2 weeks, how often have you been bothered by any of the following problems? Little interest or pleasure in doing things Feeling down, depressed, or hopeless Trouble falling asleep, staying asleep, or sleeping too much Feeling tired or having little energy Poor appetite or overeating Feeling bad about yourself, feeling that you are a failure, or feeling that you have let yourself or your family down Trouble concentrating on things such as reading the newspaper or watching television Moving or speaking so slowly that other people may have noticed. Or being so fidgety or restless that you have been moving around a lot more than usual Thinking that you would be better off dead or that you want to hurt yourself in some way Axiety: In the last 4 weeks, have you had an anxiety attack (a sudden feeling of fear or panic)? • • No (Go to question 14.3) Yes a. Has this ever happened before? b. Do some of these attacks come suddenly out of the blue that is, in situations where you don’t expect to be nervous or uncomfortable? c. Do these attacks bother you a lot or are you worried about having another attack? 176 WHY DO THEY KEEP COMING BACK? No Yes If you answered “Yes”: No Yes While answering the following questions, think about your last bad anxiety attack! a. Were you short of breath? b. Did your heart race, pound, or skip? c. Did you have chest pain or feel pressure? d. Did you sweat? e. Did you feel as if you were choking? f. Did you have hot flushes or chills? g. Did you have nausea or an upset stomach, or the feeling that you were going to have diarrhoea? h. Did you feel dizzy, unsteady, or faint? i. Did you have tingling or numbness in parts of your body? j. Did you tremble or shake? k. Were you afraid you were dying? Over the last 4 weeks, how often have you been bothered by any of the following problems? feeling nervous, anxious, on edge, or worrying a lot about different things. • • • Not at all (Go to next question) Several days More than half the days Nearly every day More than half the days Several days Over the last 4 weeks, how often have you been bothered by: Not at all If you answered Several days or More than half the days: Feeling restless so that it is hard to sit still. Getting tired very easily. Muscle tension, aches, or soreness. Trouble falling asleep or staying asleep. Trouble concentrating on things, such as reading a book or watching TV. Becoming easily annoyed or irritable. APPENDICES 177 summary Why does someone, sometimes during several years, frequently visit the general practitioner? Often, of course, because of medical problems, but sometimes the background of these frequent consultations remains unclear and the general practitioner wonders how to change this behavior. Therefore, this thesis studies (the background of) frequent attenders of the general practitioner and examines which factors lead to (long-term) frequent attendance, the relationship with the (medical) problems of the patient, the possible therapeutically approaches of frequent attenders and the impact on the workload of the general practitioner and the cost of health care. This thesis consists of three parts: 1. 2. 3. Retrospective database research on (persistent) frequent attenders; Review of the literature about interventions on frequent attenders in primary care; Prospective research of a cohort of incident frequent attenders. In chapter 1 we describe the background of this research. Anyone may have periods in their lives during which frequent help from a general practitioner (GP) is sought or needed because of a medical problem. However, when such periods exceed two or more consecutive years, not only chronic physical, but also psychosocial problems are often present. Most research on frequent attendance is cross-sectional and focusses on patients who were frequent attender during 1 year. Longitudinal studies show that only 20–30% of frequent attenders (FAs) continue to attend frequently in the consecutive year. FAs not only frequently attend their GP, but make also more use of 178 WHY DO THEY KEEP COMING BACK? other primary care and of secondary care. It is unclear whether this high use of health care may be explained by the excess morbidity these patients have. Thus, persistent frequent attendance could be seen as an easily detectable marker for underlying, often undetected, (psychosocial and psychiatric) problems. However, there are insufficient data about (the aetiology of) persistent frequent attendance, the costs of healthcare of (persistent) FAs, about effective interventions of FAs and about the influence of the GP and GP-patient communication on (persistence of) frequent attendance. It is unknown whether it would be feasible to develop a prediction rule for selecting persistent FAs using readily available information from GPs’ electronic medical records. Finally, we wanted to evaluate, using the data of a prospective cohort, whether detection and treatment of depression and anxiety of Fas could be costeffective. Because frequent attendance is mostly temporary and relates to clear and intercurrent medical problems and most somatic problems in this patient group are already dealt with in (chronic) care programs, we concentrated this thesis on persistent or repeated frequent attendance and on the specific role of psychological and social factors and GP characteristics in the aetiology of (persistence of) frequent attendance. Part 1. Retrospective database research Chapter 2. What is the best method to select FAs in a normal general practice? After considering various options, we chose a proportional definition of FAs by age and gender. Because consultation frequency depends on the age and the gender of the patient and differs between physicians, countries and regions only such a definition makes it possible to compare FAs. Using a large database of NIVEL (Netherlands institute for health services research; the first national study), we found that dividing the practice population in at least 3 age and sex groups is sufficient to reliably determine the top 10% FAs. In chapter 3 we describe the morbidity of short-term and persisting FAs and the workload of GP’s, caused by FAs using the database of the department of General Practice of the Academic Medical Center (HAG-net-AMC) This database contains data of 28.860 adult patients of 5 health centres. We found that of all Fas during one year (1yFAs), 15.4 % continued to frequently attend during 3 consecutive years (1.6% of all enlisted patients). GP’s spend 4 times more consultations on FAs and 5 times more on persistent FAs (pFAs, FAs during 3 years). Compared with non-FAs, FAs, and in particular pFAs, consume more health care and are diagnosed not only with more somatic diseases but especially more social problems, psychiatric problems and medically unexplained physical symptoms. Prevalence rates of chronic somatic illnesses vary less than psychosocial problems between non-FAs and (persistent) FAs. SUMMARY 179 Chapter 4. Besides already known morbidity (persistent) FAs may have hidden disorders. Also Fas, and in particular persistent Fas, increase GPs’ workload considerably. Therefore, it seems reasonable and efficient to target diagnostic assessment and intervention at patients with a high probability of becoming a persistent FA. But is it possible to predict which FAs are likely to continue to frequently visit the GP? Therefore, in a historic 3-year cohort study, we aimed to develop a prediction rule for selecting persistent FAs using readily available information from GPs’ electronic medical records. We used data on 28,860 adult patients from 2003 to 2005. Out of 3045 1-year FAs 470 (15.4%) became pFA. With the present indicators our rule performs modestly in selecting those at risk of becoming pFAs (area under the receiver operating characteristics curve was 0.67; 95% confidence limits 0.64 and 0.69). More information or complementary diagnostic tests seem needed to construct a rule with sufficient performance for efficient risk stratification in clinical trials. In chapter 5 we validated the prediction rule developed in chapter 4 in another region and period. We applied the existing model to a later time frame (2009-2011) in the original derivation network (temporal validation) and to patients of another network (SMILE Eindhoven; 2007-2009, temporal and geographical validation). Model improvement was studied by adding three new predictors. Finally, we developed a prediction model on the three data sets combined (N=12,539). We concluded that external validation confirmed that persistent frequent attenders can be identified moderately well using data solely from patients’ electronic medical records. FAs to primary care are likely to cost more in primary care than their nonfrequently attending counterparts. But how much is spent on specialist care of FAs? In chapter 6 we describe the healthcare expenditures of (persistent) FAs and test the hypothesis that additional costs can be explained by FAs’ combined morbidity and primary care physicians’ characteristics. We linked the pseudonymised clinical data of 16,531 patients from 39 general practices to healthcare insurer’s reimbursements data. Main outcome measures were all reimbursed primary and specialist healthcare costs between 2007 and 2009. Primary care physicians’ characteristics were collected through administrative data and a questionnaire. We concluded that FAs of primary care give rise to substantial costs not only in primary, but also in specialist care that cannot be explained by their (known) multimorbidity. Primary care physicians’ working styles appear not to explain these excess costs. The mechanisms behind this excess expenditure remain to be elucidated. 180 WHY DO THEY KEEP COMING BACK? Part II. Review of the literature about interventions on frequent attenders in primary care In Chapter 7 we analyzed which interventions are effective in influencing morbidity, quality of life and healthcare utilization of FAs in primary care. We performed a systematic literature search for articles describing interventions on FAs in primary care. Outcomes were morbidity, quality of life and use of health care. We identified 5 RCT’s of good quality. Three RCTs used frequent attendance to select patients at risk for distress, major depression and anxiety disorders and applied psychological and psychiatric interventions. Two of them found more depression-free days and a better quality of life after treating depression in FAs. No other RCT found any positive effect on morbidity or quality of life. Two RCTs studied an intervention which focused on reducing frequent attendance. No intervention significantly lowered attendance. Due to the difference in study settings and the variation in methods of selecting patients, meta-analysis of the results was not possible. Concluding we did find indications that frequent attendance might be a sign of as yet undiagnosed depression and that treatment of this depression might improve the symptoms and the quality of life of depressed FAs. We found no evidence that it is possible to influence healthcare utilization. Part III. Prospective cohort research Chapter 8. Persistent FAs frequently suffer from multimorbidity and often have many (undiscovered) psychosocial problems. Assuming that somatic problems are treated appropriately, profit could be achieved in the detection and treatment of psychosocial disorders. Better understanding of the aetiology of persistent frequent attendance could help to develop more targeted prevention. However, it is unknown what the influence of psychosocial factors and general practitioner characteristics is on persistence of frequent attendance. In a prospective cohort study with a follow-up of 2 years in 623 incident FAs in 2009, we used multilevel ordinal logistic regression analysis with 0, 1 or 2 years FB as the dependent variable and demographic, somatic, and general practitioner factors as confounders. We concluded that panic, generalized anxiety, life events, illness behavior and lack of ‘mastery’ is independently associated with persistence of frequent attendance. Prevention should focus on effective treatment of these factors. In chapter 9 we evaluated whether systematic detection and treatment of depression and anxiety after 1 and 2 years of frequent attendance might be cost-effective in comparison with usual care. We applied a Markov model to simulate the course SUMMARY 181 of a cohort of 10,000 1yFAs over a period of 5 years. Main outcomes were years of not being a FA without depression or anxiety, and Quality-Adjusted Life-Years (QALYs). We simulated 25 treatment scenarios with treatment effects expressed as risk reductions of 10%-40%. In some scenario’s spillover effects on psychiatric morbidity and frequent attender status were modeled. Uncertainty was estimated using Monte Carlo simulation (1,000 simulations). We concluded that systematic diagnostic assessment and treatment of depression and anxiety in FAs is not cost-effective in comparison with usual care, unless large spillover effects are assumed. In chapter 10 we summarize our results. First we elaborate on the strengths and limitations of our research. Secondly, we describe the literature on this topic. It is striking that, although FAs are mainly studied in countries with a similar health care system (enlisted patients), frequent attendance has only once previously been studied in a scientific study in the Netherlands. Screening and treatment programs that use frequent attendance as a ‘fishing pond’ for other illnesses (depression, somatoform disorders , but not anxiety) often have disappointing results, probably because this study design denies the multi-causal causes of frequent attendance. In the literature we found only one RCT of good quality that reduces the use of health care by FAs. In this (Spanish) research GPs analyzed FAs together and determined a targeted individual approach for each FA. Based on the results of the aetiological study (Chapter 8), we might possibly prevent some of the persistent frequent visits and costs of health care by diagnosing and treating anxiety (panic and generalized anxiety) and inadequate coping style of FAs. As long as it is not clear whether a systematic care program for FAs might be (cost) effective, we propose to identify FAs in the electronic medical file (flags) and treat them individually. This thesis advocates giving less attention to care programs focused on one single disorder (often of low prevalence) and to focus on patients, such as (persistent) FAs that can benefit optimally from the personal, continuous and comprehensive care of the GP. As described in this thesis the GP spends almost 40% of his time on FAs! Further research should focus on the background of persistent frequent attendance, to the significance of the problem list as a proxy for FA’s morbidity and to the influence of the GP on the frequency of patient consultation. A Randomized Clinical Trial will have to decide whether and which intervention might be (cost) effective in improving the quality of life and reduce health care consumption of FAs. Let’s face the frequent attender not as heart sink but as a challenge for GP care! Frequent attenders deserve better care, not more care! 182 WHY DO THEY KEEP COMING BACK? SUMMARY 183 samenvatting Waarom gaat iemand, soms meerdere jaren achtereen, vaak naar de huisarts? Meestal natuurlijk vanwege medische problemen, maar soms is de achtergrond van het frequente bezoek onduidelijk en weet de huisarts niet goed hoe hierop te reageren. Dit proefschrift beschrijft (de achtergronden van) frequent bezoek aan de huisarts, onderzoekt welke factoren leiden tot langdurig frequent bezoek, de relatie met (medisch) aandoeningen, de mogelijke aanpak van (onbegrepen) frequent bezoek en de gevolgen voor de werkbelasting van de huisarts en de kosten van de zorg. Dit proefschrift bestaat uit drie delen: 1. 2. 3. Retrospectief onderzoek met behulp van gegevens van huisartsen databases; Evaluatie van de literatuur over interventies bij frequente bezoekers in de eerste lijn; Prospectief onderzoek in een cohort incidente frequente bezoekers. In hoofdstuk 1 beschrijven we de achtergrond van dit onderzoek. Bijna iedereen kent periodes waarin, vanwege een (tijdelijk) medisch probleem, vaker contact met een huisarts nodig is. Wanneer een dergelijke periode langer aanhoudt, spelen vaak niet alleen chronische lichamelijke, maar ook psychosociale problemen een rol. Het meeste onderzoek naar frequente bezoekers (FB’s) betreft dwarsdoorsnede-onderzoek en onderzoekt frequent bezoek (FB) gedurende één jaar. Langer lopende studies tonen aan dat slechts 20-30 % van de FB’s gedurende 1 jaar vaak blijven komen in het daarop volgende jaar. FB’s bezoeken niet alleen hun huisarts vaak, maar maken ook meer gebruik van andere eerste- en tweedelijns zorg. Het is 184 WHY DO THEY KEEP COMING BACK? echter onduidelijk in hoeverre dit meer gebruik van de gezondheidszorg door FB’s verklaard kan worden door ziekten en aandoeningen die zij nu eenmaal hebben of dat hier sprake is van overgebruik. Langdurig FB is mogelijk een marker voor onderliggende, nog onopgemerkte, psychosociale problematiek. Kennis ontbreekt over (de etiologie van) langdurig FB, de kosten die (langdurige) FB’s maken in de eerste, maar ook in de tweede lijn, en over mogelijke effectieve behandelingen. Ook wilden we onderzoeken of de huisarts met gegevens, beschikbaar in het patiëntendossier, zou kunnen voorspellen welke FB’s vaak zullen blijven komen en wat de invloed is van huisarts kenmerken en huisarts - patiënt communicatie op (langdurig) FB. Tot slot wilden we met behulp van de gegevens van een prospectief cohort van FB’s evalueren of opsporen en behandelen van depressie en angst bij FB’s kosten-effectief zou kunnen zijn. Omdat FB meestal van tijdelijke aard is en omdat de meeste somatische problemen al behandeld worden in (chronische) zorgprogramma’s, concentreerden we dit proefschrift op langdurig frequent bezoek en met name op de specifieke rol van psychologische en sociale factoren en huisartskenmerken in de etiologie van (aanhoudend) frequent bezoek. Deel 1. Retrospectief onderzoek met behulp van gegevens van huisartsen databases Hoe kun je FB’s in een normale huisartsenpraktijk het best selecteren? Na bestudering van de verschillende mogelijkheden, kiezen we in hoofdstuk 2 voor een proportionele definitie van (langdurige) FB’s per leeftijd en geslacht. Omdat de bezoekfrequentie afhangt van de leeftijd en geslacht van de patiënt en verschilt per arts, land en regio, maakt alleen een proportionele definitie het mogelijk om FB’s (inter)nationaal te vergelijken. Met behulp van een database van het NIVEL (Nederlands instituut voor onderzoek van de gezondheidszorg; de tweede nationale studie) stelden we vast dat het voldoet om het patiëntenbestand van een huisartspraktijk in 3 leeftijdsgroepen per geslacht te verdelen om betrouwbaar de top 10 % FB’s te kunnen bepalen. In hoofdstuk 3 beschrijven we de aandoeningen van kortdurende en langdurige FB’s en de werkbelasting van de huisartsen door FB’s met behulp van de database van de afdeling huisartsgeneeskunde van het AMC (Hag-net-AMC). Deze database bevat gegevens van 28.860 volwassen patiënten van 5 gezondheidscentra. We vonden dat van alle FB’s gedurende 1 jaar (1jFB’s), 15,4 % frequent bleef komen gedurende 3 opeenvolgende jaren (1,6 % van alle ingeschreven patiënten). FB’s “kostten” de huisarts 4 keer zoveel consulten als zijn niet frequent komende patiënten. De langdurige FB’s (FB’s gedurende 3 jaar) gebruikten 5 maal zoveel consulten. In vergelijking met niet frequent komende patiënten hebben FB’s en vooral langdurige FB’s aanzienlijk meer sociale problemen, gevoelens van angst, verslavingsgedrag SAMENVATTING 185 en medisch onverklaarde lichamelijke klachten. FB’s hebben ook meer chronische somatische ziekten dan niet frequent komende patiënten, maar deze verschillen tussen FB’s en niet-FB’s zijn kleiner dan bij psychosociale aandoeningen. Hoofdstuk 4. Naast al bekende aandoeningen hebben (langdurige) FB’s mogelijk nog andere, onbekende (medische) problemen. Ook verhogen FB’s de werkbelasting van hun huisarts aanzienlijk. Het lijkt daarom verstandig en efficiënt om diagnostiek en preventie te richten op patiënten met een hoge kans op langdurig frequent bezoek. Maar kun je voorspellen welke patiënten vaak zullen blijven komen? Om dit te onderzoeken probeerden we, in een historische cohortstudie, vast te stellen welke informatie uit het elektronische medische dossier (EMD) voorspelt wie langdurig frequent zal blijven komen. We gebruikten de gegevens van 28.860 volwassen patiënten in de jaren 2003-2005. Van de 3.045 1jFB’s werden er 470 (15.4 % ) een langdurige frequente bezoeker. Met de huidige EMD gegevens selecteerde ons predictiemodel de patiënten die het risico lopen om langdurige frequente bezoeker te worden slechts matig (oppervlakte onder de curve 0,67; 95% betrouwbaarheidsinterval 0,64-0,69). Meer patiëntinformatie of aanvullende diagnostische tests lijken nodig om voldoende betrouwbaar langdurig frequent bezoek te kunnen voorspellen. In hoofdstuk 5 hebben we het predictiemodel, zoals beschreven in hoofdstuk 4, gevalideerd in een andere regio en periode. We pasten het bestaande model toe in een later tijdvak (2009-2011) in het oorspronkelijke netwerk (temporele validatie) en bij patiënten van een ander huisartsbestand (SMILE Eindhoven; 2007-2009, temporele en geografische validatie). We bestudeerden tevens of het toevoegen van drie nieuwe voorspellers mogelijk ons predictiemodel zou kunnen verbeteren. Tot slot, bouwden we een nieuw model op basis van de drie gecombineerde datasets (N = 12.539 ). Externe validatie bevestigde dat met de huidige gegevens uit het elektronische medische dossier langdurige FB’s slechts matig kunnen worden geïdentificeerd. FB’s maken waarschijnlijk meer kosten in de eerste lijn dan niet-FB’s. Maar hoeveel geld wordt besteed aan specialistische zorg van FB’s? In hoofdstuk 6 beschrijven we de uitgaven aan gezondheidszorg door FB’s en testen we de hypothese dat deze extra kosten kunnen worden verklaard door aandoeningen van deze FB’s en door kenmerken van hun huisarts. Wij koppelden de gepseudonimiseerde klinische gegevens van 16.531 patiënten aan de declaratiegegevens van een zorgverzekeraar. Belangrijkste uitkomstmaten waren alle vergoede kosten voor de 1e en 2e lijnsgezondheidszorg tussen 2007 en 2009. Kenmerken van de huisartsen werden verzameld door middel van administratieve gegevens en een vragenlijst. We concludeerden dat FB’s aanzienlijke kosten maken, niet alleen in de eerste, maar 186 WHY DO THEY KEEP COMING BACK? ook in de tweede lijn, die niet kunnen worden verklaard door, al bekende, medische problematiek. Ook werkstijl en kenmerken van de huisartsen lijken deze extra kosten niet te verklaren. De mechanismen achter deze extra uitgaven moeten nog worden opgehelderd. Deel II: Evaluatie van de literatuur over interventies bij frequente bezoekers in de eerste lijn In hoofdstuk 7 analyseerden we welke interventies de morbiditeit, de kwaliteit van leven en het gezondheidszorggebruik van FB’s beïnvloeden. We deden een systematisch literatuuronderzoek naar artikelen die interventies beschrijven bij FB’s in de eerste lijn. Uitkomstmaten waren morbiditeit, kwaliteit van leven en het gebruik van de gezondheidszorg. We identificeerden 5 RCTs van goede kwaliteit. Drie RCT’s gebruikten FB’s om patiënten te selecteren met een verhoogd risico voor distress, depressie en angststoornissen. Deze RCT’s pasten psychologische of psychiatrische interventies toe. Van deze drie vonden twee RCT’s meer depressie-vrije dagen en een betere kwaliteit van leven na behandeling van depressie van FB’s. Twee RCT’s bestudeerden een interventie die gericht was op het verminderen van het aantal consulten. Geen enkele interventie verlaagde dit aantal. Door verschillen in de setting van de studies en de selectie van patiënten was meta-analyse van de resultaten niet mogelijk. Concluderend vonden we aanwijzingen dat FB’s vaak een nog niet gediagnosticeerde depressieve stoornissen hebben en dat de behandeling van deze klachten de depressieve symptomen en de kwaliteit van leven van depressieve FB’s kan verbeteren. We vonden geen bewijs dat het mogelijk is om het gebruik van gezondheidszorg te beïnvloeden. Deel III Prospectief cohort onderzoek Hoofdstuk 8. Langdurige FB’s lijden vaak aan multimorbiditeit en hebben veel (onontdekte) psychosociale problematiek. Ervan uitgaande dat patiënten somatisch adequaat worden behandeld, zou winst behaald kunnen worden in het opsporen en behandelen van psychosociale aandoeningen. Beter begrip van de etiologie van langdurig FB zou kunnen helpen bij het ontwikkelen van gerichtere preventie. Wat is de invloed van psychosociale factoren en huisartskenmerken op langdurig frequent huisartsbezoek? In een prospectief cohort onderzoek met een follow-up van 2 jaar bij 623 incidente FB’s in 2009 gebruikten we een multilevel ordinale logistische regressieanalyse met 0, 1 of 2 jaar FB als afhankelijke variabele en demografische, somatische en huisartsfactoren als confounders. SAMENVATTING 187 We concludeerden dat paniek, gegeneraliseerde angst, levensgebeurtenissen, ziektegedrag en geringe ‘mastery’ onafhankelijk zijn geassocieerd met persisterend FB. Preventie van langdurig FB zou zich kunnen richten op effectieve behandeling van deze factoren. In hoofdstuk 9 onderzochten wij of systematische opsporing en behandeling van depressie en angst na 1 en 2 jaar FB kosteneffectief zou kunnen zijn in vergelijking met gebruikelijke zorg. We gebruikten een Markov model om de loop van een cohort van 10.000 1jFB’s over een periode van 5 jaar te simuleren. Belangrijkste uitkomstmaten waren jaren van niet frequent bezoek zonder depressie of angst en Quality-Adjusted Life-Years (QALY’s). Wij simuleerden 25 verschillende behandelscenario’s waarbij de effecten werden uitgedrukt als risicoreducties van 10% - 40%. In sommige scenario’s werd verondersteld dat behandeling van depressie effect zou hebben op angst en vice versa en op FB (spillover effecten). Onzekerheid werd geschat met behulp van Monte Carlo simulatie (1000 simulaties van 10.000 1jFB’s). We concludeerden dat systematische diagnostiek en behandeling van depressie en angst bij FB’s niet kosteneffectief is in vergelijking met gebruikelijke zorg tenzij grote spillover effecten aanwezig zouden zijn. In hoofdstuk 10 worden de resultaten van dit proefschrift samengevat en beschrijven we allereerst de sterke punten en de beperkingen van ons onderzoek. Daarna beschrijven we de literatuur over dit onderwerp. Hierbij valt op dat, hoewel FB’s vooral bestudeerd zijn in landen met een vergelijkbaar zorgsysteem (inschrijving op naam), FB in Nederland slechts een maal eerder onderwerp is geweest van een wetenschappelijke studie. Screening- en behandelprogramma’s die FB gebruiken als visvijver ter opsporing van andere klachten (depressie, somatoforme stoornissen, maar niet angst) hebben vaak teleurstellend weinig resultaat, waarschijnlijk omdat deze opzet de multicausale genese van FB miskent. In de literatuur vonden we slechts één goed onderzoek dat het gebruik van zorg door FB’s vermindert. In dit (Spaanse) onderzoek analyseerden huisartsen samen FB’s waarna een individuele aanpak werd vast gesteld. Gezien de resultaten van de etiologische studie (hoofdstuk 8), zou mogelijk een deel van het langdurig frequent bezoek voorkomen kunnen worden en de kosten verlaagd kunnen worden door bij FB’s diagnostiek te doen naar angst (paniek en gegeneraliseerde angst) en matige coping stijl (mastery) en door de aldus aangetoonde aandoeningen te behandelen. Zolang niet duidelijk is of een systematisch zorgprogramma voor FB’s (kosten) effectief is, stellen wij voor FB’s te ‘ruiteren’ en individueel te behandelen. 188 WHY DO THEY KEEP COMING BACK? Dit proefschrift pleit ervoor om minder aandacht te geven aan zorgprogramma’s gericht op één enkele (vaak laag prevalente) aandoening en de huisartsenzorg meer te richten op patiënten zoals (langdurige) FB’s die meer kunnen profiteren van de persoonlijke, continue en integrale zorg van de huisarts. De huisarts besteedt immers circa 40% van zijn consult tijd aan FB’s! Verder onderzoek moet worden gedaan naar de achtergrond van langdurig frequent bezoek, naar de betekenis van de Probleem Lijst als weergave van de aandoeningen van de FB’s en naar de invloed van de huisarts op de bezoekfrequentie van FB’s. Een Randomized Clinical Trial zal moeten vaststellen of en welke interventie (kosten) effectief is in het verbeteren van de kwaliteit van leven en het verlagen van de zorgconsumptie van FB’s. De frequente bezoeker moet meer een uitdaging worden voor de huisarts. Hij of zij heeft niet méér zorg nodig, maar bétere zorg! SAMENVATTING 189 dankwoord Dankwoorden in proefschriften zijn tricky things! Voor je het weet vergeet je iemand en beschadig je een (lange en gewaardeerde) samenwerking. Dus voor ik begin: Neem me niet kwalijk als ik iemand vergeten ben. Ik beroep me nu al op mijn (oude) geheugen! Laten we bij het begin beginnen. In 2003 was ik met good old Hans Grundmeijer ergens aan de borrel (Desmet?). We spraken over wat het (huisartsen) leven ons al had gebracht en wat we nog wilden doen. Na het (mede)opzetten van een gezondheidscentrum (Reigersbos in Amsterdamzuidoost), een huis verbouwen, een goede echtgenoot proberen te zijn, het (mede) opvoeden van kinderen, een huisarts zijn, mede managen van een centrum en een overkoepelende organisatie, coassistenten en aio’s begeleiden, onderzoek van andere mogelijk maken etc. , vond ik, enigszins beneveld door de drank, dat het tijd was voor nieuwe uitdagingen! En ….Hans vond dit natuurlijk een prima idee! Een nieuwe baan was geen optie (ik wilde huisarts blijven en waar vind je een leuker centrum met zulke goede collega’s?). Dus werd het onderzoek doen. We brainstormden over een goed onderwerp (“Wat puzzelt je in je vak?”) en Hans daagde me uit om hierover door te denken. Nu onder genot van koffie bleek aan zijn keukentafel dat mijn gekozen onderwerp (frequente bezoekers van de huisarts) internationaal al veel was bestudeerd, maar dat Nederlandse gegevens ontbraken. Niet zo geschikt dus, leek, in tijden dat internationaal publiceren de norm is. Hans, bedankt voor die eerste ferme en onmisbare stoot in deze richting! 190 WHY DO THEY KEEP COMING BACK? Maar vervolgens vroeg Henk (van Weert) of ik mee wilde werken aan het Apollo-D onderzoek dat hij met Aart Schene en Eric Ruhé had opgezet. Het betrof een groot onderzoek, samen met Nijmegen, over het screenen van hoog risicogroepen op depressie. Eén van de hoog risicogroepen betrof frequente bezoekers. Ik zou dat onderdeel (mede) gaan trekken. Hoewel ik veel heb geleerd in die tijd (van Jochanan Huijser, Aart Schene, Eric Ruhé en Henk van Weert) en het ook een gezellige tijd was op het kamertje in het oude psychiatrie gebouw (met Karin Wittkampf, Kim Baas en onderzoeksassistente Judith Kosman) bleek na enige tijd dat de koek van Apollo-D gegeten zou worden door de fulltime promovendi uit Amsterdam en Nijmegen. Ik koppelde mijn 5e wiel aan de wagen noodgedwongen af. Een poging in 2005 om, samen met Aart, subsidie te krijgen van het Nationaal Fonds Geestelijke Volksgezondheid voor een apart vervolg onderzoek mislukte helaas. Karen, Kim en Judith, Henk, Aart, Eric en Jochanan bedankt voor die leerzame en gezellige tijd! Als langdurig lid van de stuurgroep HAG-net-AMC kende ik, buiten Henk van Weert, al langer Gerben ter Riet, Henk Brouwer en Jacob Mohrs. Zij wilden me gelukkig helpen om de ideeën die er lagen uit te werken. Zij waren als geen ander thuis in de rijke, meerjarige bestanden van het HAG-net-AMC. Deze ‘mannengroep’ leidde, mede, tot 4 artikelen ( hoofdstuk 2, 3,4 en 7). In 2008 heeft Gerben ter Riet het initiatief genomen om een aanvraag voor ZonMW, programma alledaagse ziekten, te schrijven voor een vervolg onderzoek. Dit werd gehonoreerd en ik kon nu voor één dag in de week aan de slag als onderzoeker in het kader van het PERFACTIO project (acroniem voor: PERsistent Frequent Attenders risk faCtors and Treatment optIOns). Officiële start 01 12 2008. Onderzoek doen is teamwork!: Er werd een begeleidingsgroep geformeerd (Henk van Weert, Aart Schene, Marcel Dijkgraaf, Gerben ter Riet) en een werkgroep (Gerben ter Riet, Henk Brouwer, Jacob Mohrs en Diana Toll). Later hebben Marcel en Diana ons verlaten en hebben we succesvol samenwerking gezocht met de afdeling gezondheidswetenschappen van de VU (Judith Bosmans). Leo Beem versterkte ons team statistisch. Gerben, Henk B en Jacob bedankt voor jullie stimulans, hulp en kennis van de hag-net bestanden en SPSS! Het waren stimulerende, gezellige en productieve bijeenkomsten op jullie kamers. De (secretariële en onderzoeks) ondersteuning was al die jaren in de prima handen van Alice Karsten, Gerda van Zoen en Nienke Buwalda. Alice, Nienke en Gerda DANKWOORD 191 enorm bedankt voor jullie vele hulp en spandiensten. Ik wil een paar personen apart noemen. Allereerst de ‘mannengroep’, het ‘dagelijks bestuur’ van Perfactio: Gerben, jij was al deze jaren de stimulerende, onvermoeibare onderzoeksleider. Ik ben je heel veel dank verschuldigd voor het in goede banen houden van Perfactio. Je grote epidemiologische en onderzoekstechnische kennis waren onmisbaar. Maar vooral ook de kameraadschap die ik al die jaren bij je gevoeld heb, zal ik niet snel vergeten. We hebben het toch maar geflikt! Henk B, zonder mijn fijne contacten met jou, al vanuit de “transitie”tijd, zou ik waarschijnlijk niet met dit onderzoek zijn begonnen. De manier waarop jij mijn (klinische) vragen uit de hag-net bestanden wist te toveren hebben mij vaak blij verrast. Heel wat uren hebben we samen op je kamer aan extracties gewerkt met dit kloeke boek als resultaat. Enorm bedankt! Jacob Mohrs, jij was de onverstoorbare ‘diesel’ van Perfactio. Jij maakte bestanden, bouwde een onderzoeksdatabase en onderhield de contacten met Saxion en de drukker over de (scan bare) vragenlijsten. Ik kon altijd op je bouwen en bij je terecht voor hulp of een gezellig praatje. Heel erg bedankt! Leo Beem, helaas hebben we, door je overlijden, afscheid van je moeten nemen. Je was onnavolgbaar met cijfers, maar je dikke pak output leverde altijd toch een bruikbare tabel op. Het ga je goed daarboven! Koos Zinderman, gelukkig was jij bereid om Leo’s taken over te nemen. Heel veel bewondering heb ik voor de rustige manier waarop jij, tussen je drukke werkzaamheden door, tijd vrij maakte voor ons onderzoek. Jij was als geen ander thuis in onze database! En …ik snap nu iets meer van de wondere achtergronden van genen en quality of life! Maar ook het ‘algemeen bestuur’ van de grote begeleidingsgroep was groots: Allereerst mijn (co)promotores: Henk vW, we kennen elkaar al lang als collega huisarts en het was even wennen aan deze ‘change of position’. Dit heeft niet lang geduurd en ik heb voluit geprofiteerd van je grote huisartsgeneeskundige kennis en onderzoekservaring. Ik kon altijd bij je terecht voor een goed advies, maar ook om even te bomen over de politiek, de gezondheidszorg, de GAZO en wat niet meer. Aart, ik leerde je kennen tijdens Apollo en sindsdien ben je bereid geweest om je grote psychiatrische expertise in te zetten voor de frequente bezoekers van de huisarts. Heel erg bedankt voor deze langjarige interdisciplinaire inzet en hulp! Erg bedankt ook voor je altijd snelle en zeer to-the-pointe commentaar op stukken en concept artikelen. Judith, jij was onze kenner van kosten en centen. Als VU’er en als enige vrouw had je het soms moeilijk met de haantjes van het AMC, maar we hebben enorm kunnen 192 WHY DO THEY KEEP COMING BACK? profiteren van je kennis op het gebied van kosten(effectiviteits)onderzoek. Bedankt voor je inzet, het meedenken, je prima commentaar op concepten en het (mee) schrijven! Gerben heb ik al gememoreerd! De andere leden van de leescommissie, prof. H. v.d. Horst, prof. T. Lagro, prof. K. Stronks, prof. M. Maas, prof. J.de Haes en dr. Eric Ruhé, wil ik hartelijk danken voor het lezen en beoordelen van mijn proefschrift en de bereidheid om tijd vrij te maken voor de promotie. Beste collega’s van het gezondheidscentrum Reigersbos, jullie zijn natuurlijk onmisbaar geweest. Ten eerste hebben jullie mij mede de inspiratie gegeven voor dit onderzoek, maar ook was onze samenwerking altijd de fijne, stabiele en gezellige rots in de branding. Ik geloof niet dat jullie verder veel “last” hebben gehad van mijn andere “baan”, misschien ook wel baten. Heel erg bedankt voor jullie collegialiteit en vriendschap! Zonder de medewerking van de huisartsen en patiënten van het Hag-Net-AMC was dit alles niet mogelijk geweest. Heel erg bedankt voor jullie moeite! Tot slot mijn thuisfront. Jullie waren, op de achtergrond, erg belangrijk voor het slagen van dit project. Ik besef heel goed dat ik enigszins heb geparasiteerd op jullie en dat ik jullie en onze vrienden heb verwaarloosd. Dat moet maar weer snel veranderen, want zonder jullie was ik nergens en was dit proefschrift trouwens nooit gelukt. Lieve vrienden en buren, dankzij jullie ging het leven gewoon door en was er ook de zo nodige afleiding. Bedankt voor jullie vriendschap! Lieve, lieve Chris, heel erg bedankt voor je liefde, je geduld, maar ook voor de (psychiatrische) inspiratie die je me heel vaak hebt gegeven en de vele hulp bij deze promotie. Er breken nu langzamerhand andere tijden voor ons aan! Ik hoop nog veel gelukkige jaren samen met jou te beleven! Lieve Laura en Thomas, paranimfen, maar vooral dochter en zoon, bedankt voor het vormgeven van dit boekje (Laura) en het organiseren van de feestelijkheden (Thomas), jullie vele hulp bij de promotie, maar vooral voor jullie inspirerende liefde en gezelligheid. Chris, Laura, Thomas en Clara, ik hoop nog lang veel pittige discussies met jullie aan tafel te voeren! En nu maar hopen dat ik zelf nooit een frequente bezoeker van de (huis)arts zal worden! DANKWOORD 193 curriculum vitae Frans T.M.Smits werd geboren op 21 december 1950 te Breukelen. Na het behalen van zijn Gymnasium-bèta-diploma aan het Sint Bonifatius College te Utrecht in 1969, studeerde hij geneeskunde aan de Radboud Universiteit Nijmegen. Na het behalen van het artsexamen in 1977, werkte hij tot 1978 als arts-assistent in het psychiatrisch ziekenhuis, huize Padua, te Boekel. Van 1978 tot 1979 volgde hij de specialisatie tot huisarts aan de Radboud Universiteit Nijmegen. Van 1979 tot 1981 werkte hij als verpleeghuisarts in verpleeghuis Dekkerswald te Groesbeek. Tevens bereidde bij in die jaren mede de start voor van een nieuw gezondheidscentrum in Amsterdam. In 1982 startte hij een huisartsenpraktijk in het Gezondheidscentrum Reigersbos, te Amsterdam-zuidoost. Hij was en is bestuurlijk actief binnen de stichting Gezondheidscentra zuidoost, de afdeling huisartsgeneeskunde AMC en regionale/stedelijke organisaties. Hij was opleider van coassistenten en is opleider van artsen die zich specialiseren tot huisarts. In 1984 participeerde hij in het transitieproject, een registratie project, van prof. Henk Lamberts. In 1994 maakte hij deel uit van de congrescommissie van het jaarlijkse NHG-congres (”Huisarts en zinvol handelen- tussen teveel en tekort doen”). Van 2005 tot 2007 participeerde hij in de onderzoeksgroep van het Apollo-D project, een onderzoek van de afdelingen huisartsgeneeskunde en psychiatrie van de universiteit van Amsterdam en van de Radboud Universiteit Nijmegen naar een screeningsprogramma van depressieve stoornissen bij hoog risicopatiënten in de huisartspraktijk. Van 2007 tot 2009 deed hij aan de afdeling huisartsgeneeskunde van het Academisch Medisch Centrum, universiteit van Amsterdam 194 WHY DO THEY KEEP COMING BACK? onderzoek naar frequente bezoekers van de huisarts. Van 2009-2014 was hij als huisartsonderzoeker verbonden aan het PERFACTIO project, een door ZonMw gesteund onderzoek naar de risicofactoren voor en de behandelmogelijkheden van (langdurig) frequent bezoekers aan de huisarts. Frans is gehuwd met Chris E. Folkers en heeft twee kinderen, Laura (1985) en Thomas (1988). CURRICULUM VITAE 195 PhD portfolio 196 PhD period: December 2008 - December 2013 PhD-supervisors: Dr. Gerben ter Riet Prof. Dr. H.C. van Weert Prof. Dr. A.H.Schene WHY DO THEY KEEP COMING BACK? Overview of PhD related activties PhD Training Year Workload(hrs) Cursus ‘Doelmatigheids onderzoek: methodes en principes’ (VU) 2010 21 Presentation hag-net-amc meeting 2009 4 Presentation at the EGPRN conference, Bertinoro, Italy (European General Practice Research Network) 2009 20 Poster presentation at the NHG wetenschapsdag 2010 4 Presentation at the NHG wetenschapsdag 2012 10 Presentation for colleagues of the st. Gazo 2012 4 Presentation at the Achmea congress 2013 10 Presentation at the NHG wetenschapsdag 2014 10 WONCA Europe Regional Conference, Istanbul 2008 21 the EGPRN conference, Bertinoro, Italy 2009 20 NHG-wetenschapsdag 2010 6 NHG conference 2011 6 NHG-wetenschapsdag 2012 6 Achmea conference 2013 6 NHG wetenschapsdag 2014 6 focusgroup of the gezondheidsraad 2010 2 Commentary in Br J Gen Pract. 2010 Apr;60(573):293-4 2010 2 Review article ‘Journal of Affective Disorders’: Published as: J Affect Disord. 2011 Jun;131(1-3):428-32 The mental health of doctor-shoppers: experience from a patient-led fee-for-service primary care setting) 2010 4 Review article Scandinavian Journal of Primary Health Care: 2013 4 Letter to the editor (NRC) 2013 2 Response to Des Spence (BMJ) (https://cealu11fn98fef.sec.amc.nl/content/348/bmj. g208?tab=responses) 2014 3 Presentations (Inter) national conferences Other activities PHD PORTFOLIO 197 list of publications Publications 198 1. Contributions to: Gezondheidszorg in Nederland. Sun, Nijmegen, 1973. 2. Smits F.T: Etniciteit als contextuele factor; Huisarts en Wetenschap 01/2002; 45(2) 813-813 3. Smits F.T, Mohrs JJ, Beem EE, Bindels PJ, van Weert HC: Defining frequent attendance in general practice. BMC Fam Pract 2008,9:21. 4. Smits F.T, Wittkampf KA, Schene AH, Bindels PJ, van Weert HC. Interventions on frequent attenders in primary care. A systematic literature review. Scand.J Prim. Health Care 2008;26(2):111-6. 5. Wittkampf KA, van ZM, Smits FT, Schene AH, Huyser J, van Weert HC. Patients’ view on screening for depression in general practice. Fam Pract 2008; 25(6):438-444. 6. Wittkampf KA, Van Zwieten M, Smits FT, Schene AH, Huyser J, Van Weert HC. Wat denken gescreende patiënten met een depressie over hun diagnose? Huisarts Wet 2009;52(6):274-80. 7. Smits FT, Brouwer HJ, Ter Riet G, van Weert HH. Epidemiology of frequent attenders: a 3-year historic cohort study comparing attendance, morbidity and prescriptions of one-year and persistent frequent attenders. BMC. Public Health 2009;9:36. WHY DO THEY KEEP COMING BACK? 8. Smits FT, Brouwer HJ, van Weert HC, Schene AH, Ter Riet G. Predictability of persistent frequent attendance: a historic 3-year cohort study. Br.J Gen.Pract. 2009 Feb;59(559):e44-50. 9. Smits F T , Brouwer H J, van Weert HC, Schene AH, ter Riet G.: Langdurig frequent bezoek aan de huisarts moeilijk te voorspellen. Huisarts en wetenschap. 2009:4:166-172 10. Frans Smits,, Henk Brouwer, Gerben ter Riet. Letter to the editor: Persistent frequent attenders.Br J Gen Pract April 1, 2010 60:293-294 11. Smits FT, Brouwer HJ, Zwinderman AH, van den Akker M, van SB, Mohrs J, Schene AH, van Weert HC, Ter RG: Predictability of persistent frequent attendance in primary care: a temporal and geographical validation study. PLoS One2013:e73125. 12. Smits FT, Brouwer HJ, Zwinderman AH, Mohrs J, Smeets HM, Bosmans JE, Schene AH, van Weert HC, Ter RG: Morbidity and doctor characteristics only partly explain the substantial healthcare expenditures of frequent attenders: a record linkage study between patient data and reimbursements data. BMC Fam Pract 2013, 14:138 13. Smits FT, ter Riet G, Bosmans J, Haroun D: Better care for frequent attenders is possible. Rapid response BMJ; http://www.bmj.com/ content/348/bmj.g208?tab=responses 14. Letter to the editor of NRC, 26 sept 2013: “Surinamers zijn helemaal niet duurder in de zorg” 15. Frans T. Smits, Henk J. Brouwer, Aeilko H. Zwinderman, Jacob Mohrs, Hugo M. Smeets, Judith E. Bosmans, Aart H. Schene, Henk C. van Weert en Gerben ter Riet: Frequente bezoekers van de huisarts maken hoge kosten in de eerste en tweede lijn. Ned Tijdschr Geneeskd. 2014;158:A7117 LIST OF PUBLICATIONS 199 200 WHY DO THEY KEEP COMING BACK?