Nine-Year Follow-Up of Danish Chronic Fatigue Syndrome (CFS) Patients:
Impact on Health, Social, Vocational, and Personal Lives
---------------------------------------------------------------------Mette Marie Andersen, MD; Henrik Permin, MD, DMSci; Frank Albrecht, Psych, PhD
- Mette Marie Andersen, Henrik Permin, and Frank Albrecht are affiliated
with the Department of Internal Medicine L, Bispebjerg Hospital, DK-2400,
Copenhagen, Denmark.
- Address correspondence to: Henrik Permin, MD, Department of Internal Medicine L, Bispebjerg Hospital, DK-2400, Copenhagen, Denmark (E-mail:
HP16@bbh.regionh.dk).
ABSTRACT
Objective
To determine quality of life (QOL) and health in Danish CFS patients 9 years
after diagnosis.
Methods
Thirty-four adults with CFS responded to questions regarding QOL at diagnosis,
and again 5 and 9 years later. At 9-year follow-up patients also responded to
questions regarding health, fatigue, use of Health Care system, alcohol and
exercise.
Results
Two patients (6%) had recovered and 3 patients (10%) had received secondary
diagnoses. Overall, there was no improvement, except with depression/anxiety.
The order of severity among disabilities remained the same. Work had the
highest disability score, followed by post-exertional malaise. Patients slept
and rested 13.6 hours a day (mean). Self-reported physical health correlated
with hours sleeping and resting. Rheumatic symptoms dominated the health
symptoms. Alcohol consumption was low, and the use of the Health Care system
was modest.
Conclusion
After 9 years QOL was the same as at diagnosis, only mental health had improved.
KEYWORDS. Chronic fatigue syndrome, 9-year follow-up, quality of life, work
disability, post-exertional malaise, mental health, hours sleeping and resting,
physical health
INTRODUCTION
Studies on the natural course of CDC-defined CFS in adults have focused
mainly on percent remission. In a systematic review, Cairns and Hotopf (1)
found a median full recovery rate of 5% (range 0-31%). Work-related
impairment with CFS was reviewed by Taylor and Kielhofner (2), who found
unemployment ranging from 35-69% and job loss ranging from 26-89%.
The present report of the natural course of CFS over 9 years is comprehensive, covering work, general health, social life, and long term
disability. It is based on self-report quality of life (QOL) data from 34
Danish CFS patients at 3 points in time (1994, 1999 and 2003). In 2003 we
also asked the patients about health-related questions.
METHODS
In 1994 the first 34 patients to be diagnosed with CFS were recruited for
this study. These patients had been seen on referral from general
practitioners by one of us (Permin), and diagnosed with CFS only if meeting
both the Holmes et al. (3) and the Fukuda et al. (4) criteria. At diagnosis
mean age was 46.4 years and mean duration of illness was 4 years. Details of
patients and methods were described earlier (5). Each patient responded to a
50-question QOL questionnaire. This instrument was re-administered 5 and 9
years later (the "Repeated Measure"). At 9 years patients also responded to a
74-question questionnaire, "Health in the Past 12 Months," covering general
health, use of the health care system and health related behaviour (the "New
Health Measure," Appen- dix A).
All patients were followed in the out-patient clinic from 1994 to 1999.
Standard medical care was provided and symptoms (sleeplessness, pains,
allergies, etc.) were treated accordingly. In 2003 patients were contacted by
phone or e-mail and sent the questionnaires by mail. One patient was too ill
to fill out the papers himself. Permin recorded his responses by telephone.
We obtained information on patients' disability benefits, pensions, and
household access to a vehicle from other sources.
Scoring of Data, Repeated Measure
Responses to scaleable questions were scored on a 3 point scale (0, 1, 3),
with zero indicating either no CFS-related disability or (on symptom-related
questions) significant improvement. The highest score represented maximum
disability or that the symptoms were much worse. Percent disability was
defined as the actual score (x100) divided by the maximum possible disability
score. Nine outcome variables were con- structed: work status, social
impairment, hours per day sleeping and resting, cognitive disability,
driving, neurological disturbances and allergies, depression/anxiety (D/A),
sexual problems, and post-exertional malaise. Maintenance of a full or part
time paid job defined normal work status. Normal functioning for other
outcome variables was defined as a score of less than 30% of maximum
disability.
Scoring of Data, New Health Measure
"CFS-Related Symptoms" (Appendix A-1) were scored on a threepoint scale as
with the Repeated Measure. Scoring of "Nature of Fatigue" was done (Appendix
A-2). For data presentation the numbers were transformed from the 1-7 scale
to a -3 - 3 scale. Disagreement with statements resulted in negative figures.
"Contact with Health Care System" was recorded as shown (Appendix A-3).
Alcohol intake was recorded in units (drinks) (Appendix A-4). Five outcome
variables were constructed: CFS-related symptoms, physical health in general,
mental health, nature of fatigue, and fitness.
Statistical Analysis
Wilcoxon Matched-Pairs Signed Ranks test (Wilcoxon) was used to compare the
outcome variables from 2003 with those from 1994. Analysis Of Variance
between groups (ANOVA) was used to correlate individual 2003 outcome variables.
Three patients with secondary diagnoses were excluded from statistics.
RESULTS
The 2003 response-rate was 100%. Two patients (6%) fulfilled criteria for
full recovery. One (male) patient was seriously ill (bed-bound for 24 hours a
day, body weight 45 kg, requiring constant care). The remaining patients
managed their own lives to varying degrees. Three patients had received
secondary diagnoses prior to 2003 (breast cancer, Addison's disease and
chronic hepatitis C). We did not exclude these 3 patients but, as already
mentioned, did not use their data in the statistical analysis.
Repeated Measure, 2003 Compared to 1994
Disability scores in Tables 1 and 2 are shown in order of decreasing severity.
1. Work: One patient worked full-time but had changed to a less
physically demanding job. Two had part-time jobs. Twenty-six
patients received disability benefit or retirement pension, while
3 were provided for by their spouse. Four patients stated they
had free-lance jobs (as writer, congregation worker, voluntary
editor and artist, respectively) but each of these also received
disability payments.
2. Post-exertional malaise: PE-malaise scored the second highest,
and was unchanged compared to 1994.
3. Sexual problems: Only 6 patients were as sexually active as before becoming ill, while 19 reported less or no sexual contact
with their partner. The majority (78.9%) were too fatigued to
have sex, while 36.8% had reduced libido, or a combination of
both. Nine patients had no partner. This was an increase compared to previous years (6 in 1999 and none in 1994 had been
alone). Information on whether loss of partner had anything to
do with reduced sexual contact was not obtained.
4. Neurological problems: Sleep problems (87.3%) scored the highest among the neurological problems.
5. Depression/Anxiety (D/A) factor: The D/A score in 2003 was
55.1%. This represented a significant improvement compared to
1994 (Wilcoxon p<0.02). Panic attacks scored especially low.
6. Cognitive disabilities: With a mean disability score of 53.4%,
the 3 items with the highest score were 'tip-of-tongue problem,'
'attention deficit while reading,' and 'forgetting names.'
7. Social impairment: 'Reduction in out-of-home entertainment'
and 'time spent on former hobbies' had the highest disability
scores.
8. Driving: Among the 21 patients having a drivers licence and access to a household car, the mean driving disability score 36.7%.
This included 9 who felt safe when driving and 12 who were
more or less afraid to drive. The score was unchanged compared
to 1999 and 1994. Two patients did not have a drivers licence.
We do not know if the disposal of a car in 11 households was
health related or for economic reasons.
9. Hours sleeping and resting: The mean number of hours sleeping
and resting was 13.6 hours a day. This (non-significant) reduction compared to 1994 was due to a reduction in sleep only.
New Health Measure, 2003
Disability scores are in order of decreasing severity, Tables 3 and 4.
10. Fitness: Twenty-four patients judged their fitness as much worse.
Six patients judged their fitness as being the same or better,
while 4 judged it somewhat worse.
11. Physical health in general: Eight patients judged their health as
being good, 12 judged it as tolerable and 14 patients judged their
physical health as poor. Patients who judged their health as poor,
slept and rested for 15.1 hours a day, while those with tolerable
health slept and rested for 14.0 hours a day. Patients who judged
their health as good slept and rested for 10.2 hours a day (mean)
(ANOVA p<0.037). Physical health showed no correlation
with fitness.
12. Mental health: Fifteen patients judged their mental health good
and 12 who judged it tolerable. The remaining 7 patients judged
their mental health to be poor. Mental health was significantly
correlated with physical health (ANOVA p<0.001).
13. CFS-related symptoms: Patients suffered from a mean of 7
symptoms among the 18 symptoms listed. Fibro-pain, fibrostiffness and joint pain had the highest symptom severity-scores;
but restless legs, tingling of hands and feet, irritable bowel,
headache, and brainfog also had high scores. Symptom score
was significantly correlated with physical health (ANOVA p<0.02),
but not with mental health. In addition, there was no significant
relationship between age and the following measures: fitness,
physical health, mental health, symptom score and sleep/rest time.
14. Nature of fatigue: Most patients agreed with the statements "fatigue prevents lasting physical performance," "physical activity
makes me fatigued" and "following physical activity I feel fatigued for a long time" (Table 4). Most patients disagreed with
the statements "inactivity makes me fatigued" and "fatigue is
worst in the morning."
New Health Measure, Other Results, 2003
Use of the health care system: Thirty patients had seen their general
practitioner and 20 patients had seen a specialist in the past 12 months.
Twelve patients had visited alternative practitioners. Ten had seen a
physiotherapist. Six had had a hospital admission. None had seen a
psychiatrist.
Alcohol: Sixteen patients (47%) never drank alcohol. Those who drank had a
mean intake of 1.76 units (drinks) of alcohol per day. In Denmark, mean adult
alcohol consumption is 2.1 drinks per day.
Exercise: Walking was the most common form of exercise, engaging 19 patients
(56%) for 2 hours or more a week. Seven patients did sports for 2 hours or
more a week, while a total of 11 patients exercised less than 2 hours a week.
Medicine for CFS Symptoms
Five patients took low doses of thyroid hormone and 3 took low doses of
corticosteroids. Thirteen patients were taking sedatives, 5 took antidepressants periodically. Fourteen took sleeping medications.
Twenty-five patients took over-the-counter medicine, mostly painkillers
(aspirin and acetaminophen), 21 patients took nutritional supplements,
including multivitamins and minerals.
DISCUSSION
This 9-year follow-up study of QOL in CFS showed that our patients as a group
had neither improved nor deteriorated since diagnosis. The relative order of
severity for the outcome variables stayed the same; the disabilities which
were most severe or disturbing at diagnosis remained so throughout. As
reported previously (5), we found full and sustained recovery in 2 patients
(6%). Three patients had received secondary diagnoses, but in each case the
specific treatment given had not improved their CFS symptoms including
fatigue. Our results underscore the chronicity of CFS and provide prognostic
guidance for physicians, patients, and their families.
The high score for work disability is probably an inevitable consequence of
the fact that most patients suffered from post-exertional (PE) malaise, with
massive needs for sleep and rest. In addition, patients needed to control the
timing of their energy expenditure, a need that is not compatible with almost
all forms of regular employment. The high score for PE-malaise supports other
studies showing PE-malaise as one of the cardinal markers of CFS (6).
Patients slept and rested for a mean of 13.6 hours a day, with sleeping
accounting for 9.0 hours. Patients reporting better health in general
reported sleeping less than those reporting poorer health, not surprising in
an illness characterized by fatigue. The mean score was also high on the
question "Do you have difficulties sleeping (either sleep much more than
before, cannot fall asleep, wake up frequently)?" Scores for night time
urination, and for nightmares, indicated that patients woke up frequently.
Almost half the patients took sleeping medication, suggesting problems with
falling or staying asleep. Most patients reported one or more kinds of sleep
problems after 9 years.
Our patients had significant problems with sexual relations with partners.
The majority attributed these problems to fatigue. Jason et al. (7) found the
presence of sexual problems indicative of CFS, whereas Vermeulen and Scholte
(8) did not find problems with sexual interest or desire with their patients.
This topic deserves more study.
Among those who felt depressed, the vast majority said they were feeling
depressed about the illness. This indicates adjustment reaction rather than
depressive illness. Only 5 patients took anti-depressive medicine in 2003,
supporting the finding by Ax et al. (9) that antidepressants are ineffective
for treatment of CFS fatigue. None of our respondents had felt the need to
see a psychiatrist over the preceding 12 months.
Statements about fatigue that scored the highest were related to physical
activity, stress, work, and concentration difficulties. The statement
"fatigue is worst in the afternoon/evening" scored high, while "fatigue is
worst in the morning" scored very low. This is another indication that
patients do not suffer from major depression.
Overall, the D/A factor improved significantly from 1994 to 2003. This
finding is supported by patients' low scores on mental health related
disability in 2003, supporting the view that mental health problems in CFS
are adjustment reactions. Over time emotional integration and coping skills
improved despite lack of physical recovery.
Our patients have been ill for many years. They probably have obtained
extensive experience with their exercise limitations. More than half of our
patients took walks for 2 hours or more a week, suggesting that walking is
not experienced as a strenuous activity. Lehman et al. (10) found that
patients who believed that the path to recovery was through limiting their
physical exertion had significantly lower depression and anxiety scores than
their counterparts. We feel confident that our patients were able to respect
their own limits and exercise as much as they possibly could. Until more
information is presented on the pathophysiology underlying PE-malaise,
recommending strenuous exercise to CFS patients is hazardous.
The disability score for cognitive abilities and memory covered a range of
problems in addition to the issue of car driving. Coping with modern traffic
demands more cognitive skills than available to most of our patients. The
driving disability score was recorded among those with a car, and some
patients may have disposed of their car because they were unable to drive it.
Scores for social impairment were also unchanged over time. Sleeping and
resting for 15 hours a day leaves little time activity or recreational
hobbies. Many patients stated they felt they lived "only half-a-life."
Almost half the patients abstained from drinking alcohol, and for those who
did drink, the mean intake was low compared to Danish adult population.
Alcohol intolerance in CFS patients was also found by Woolley et al. (11),
and may be of interest in connection with research on the pathophysiology
behind CFS.
Thirty patients had seen their general practitioner at least once during the
past 12 months. This is much below the norm for doctor visits in the Danish
population (mean=6.5 visits per year). Our high response-rate at the CFS
clinic and the low use of other health care utilities could be attributed to
the quality of the relationship of Permin with the patients, as well as to
vigorous follow-up with potential non-responders.
Our findings were based on self-report questionnaires. Such instruments have
well-known limitations, such as recency effects, faulty memory, and problems
with understanding the questions. In 2003 the vast majority of patients
stated that they experienced daily, weekly, or monthly symptoms fluctuations.
Because the focus of our instruments was current symptoms and problems, we
were unable to shed any light on this important and interesting topic. In
addition, rheumatic symptoms dominated the list of health problems troubling
our patients. We had not previously asked about these symptoms, so we do not
know whether these problems have worsened since 1994.
TABLE AND APPENDIX CAPTIONS
There are no electronic versions of the tables and the appendices available.
Table 1. Disability scores in 34 CFS patients 2003-1994
Table 2. Disability scores in 34 CFS patients 2003-1994
Table 3. Fitness, Health and CFS-related symptoms 2003
Table 4. Nature of Fatigue 2003
Appendix A. The New Health Measure. Questions regarding the past 12 months
1. Health
2. Fatigue
3. Contact with health care system
4. Alcohol & exercise
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-------(c) 2007 The Haworth Press
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