Cognitions associated with anxiety in Ménière’s disease Running head: Anxiety in Ménière’s disease School of Psychology, University of Southampton, UK Sarah. E. Kirby, PhD, and Lucy Yardley, PhD Address correspondence to: Sarah Kirby, School of Psychology, University of Southampton, Highfield, Southampton SO17 1BJ, UK. Tel: +44 (0)23 8059 2581 Fax: +44 (0)2380 594597. E-mail: Sarah.Kirby@soton.ac.uk Anxiety in Ménière’s disease 2 Abstract Objectives: The purpose of this longitudinal study was to identify cognitions associated with anxiety and maintenance of anxiety in people with Ménière’s disease. Method: At baseline participants completed the Hospital Anxiety and Depression Scale (HADS), the Revised Illness Perception Questionnaire, the Dizziness Beliefs Scale, the Fear Avoidance Beliefs Questionnaire, the Intolerance of Uncertainty Scale, and measures of demographic and illness characteristics. Participants were then randomised to no treatment or to receive one of two self-help booklets, and completed the HADS again at 3 month follow-up. Results: After controlling for symptom severity, baseline anxiety was associated with intolerance of uncertainty, fearavoidance of physical activity, the belief that dizziness would develop into a severe attack of vertigo, and several illness perception subscales (emotional representations, consequences, psychological causes, and perceived treatment effectiveness). Anxiety at follow-up was predicted by higher baseline levels of autonomic/somatic symptoms and intolerance of uncertainty, and reporting less understanding of the illness. These longitudinal relationships were found in those who did and did not receive self-help booklets. Conclusions: Our findings suggest that intolerance of uncertainty is associated with anxiety in Ménière’s disease. A controlled trial is needed to see whether anxiety might be reduced in Ménière’s disease by helping patients to tolerate and cope with uncertainty. Keywords: anxiety disorder, vestibular, Ménière’s disease, attitudes, questionnaire design. Anxiety in Ménière’s disease 3 Introduction Ménière’s disease is an incurable chronic disorder of the inner ear, characterised by recurrent spontaneous attacks of severe vertigo (a strong sense of spinning), progressive hearing loss that becomes permanent in one or both ears, a sense of fullness or pressure in the ear(s), and intermittent spells of loud tinnitus (a buzzing, ringing or roaring sound) [1]. There are close neurological links between the vestibular and autonomic systems, with the consequence that vestibular disturbance directly provokes autonomic symptoms such as nausea, pallor and sweating (as in motion sickness). Following an acute attack of vertigo, dizziness gradually diminishes as the central balance system habituates to the change in vestibular function. However, residual dizziness can still be provoked by unaccustomed movements and disorienting situations [2;3]. Autonomic symptoms can also be induced by anxiety arousal [4-6]. Both illness and anxiety provoked symptoms have the potential to create a vicious cycle of prolonged symptomatology and distress, as symptoms can be augmented by anxiety, and in turn fuel further anxiety [6-9]. Indeed, high levels of anxiety are often reported among those who experience vertigo [10-12], and elevated levels of anxiety and distress have been found in people with Ménière’s disease [13-15]. It would be helpful to be able to identify modifiable factors that are associated with anxiety in order to try to limit the exacerbation of this vicious cycle. Cognitive behavioural approaches to chronic illness suggest that cognitions about illness and its consequences are important in how people with chronic illness respond emotionally to their illness [16;17]. Therefore, if the cognitions that contribute to anxiety in Ménière’s disease can be identified, this should assist in the identification of the forms of support and therapy that are most likely to reduce Anxiety in Ménière’s disease 4 anxiety in people with Ménière’s disease. This study considers the relevance to Ménière’s disease of three groups of cognitions found to be related to anxiety among other chronic illnesses: illness perceptions, dizziness related fears and beliefs, and intolerance of uncertainty. Anxiety is likely to be related partly to realistic negative cognitions, but in chronic illness anxiety is also often related to excessive and catastrophic concerns, which may be amenable to modification. Illness perceptions [18;19] have been found to play a significant role in relation to a variety of outcomes, including anxiety [20]. Chronically ill people experience more psychological distress if they have a strong illness identity (i.e. attribute many symptoms to the illness), a stronger emotional response to illness, feel they do not understand their illness well, and believe that their illness has serious consequences, will last a long time and cannot be easily controlled [21-23]. These perceptions of illness may be common Ménière’s disease. A strong illness identity could develop if symptoms of anxiety arousal and residual dizziness were attributed to active disease. Moreover, some people with the disease may have to make significant lifestyle changes, including changing or giving up work or certain social or leisure activities, or becoming unable to drive or travel. Therefore people may well view the disease as having serious consequences, depending on the extent to which it has impacted on their family and finances, and social and occupational areas of life. The disease is incurable and treatment options are limited, as little is known about what causes the disease. Therefore people with Ménière’s disease may correctly expect their illness to be long-lasting, and may also believe that they do not understand their illness very well, and that the symptoms cannot be easily controlled. Dizziness related fears and beliefs have also been found to contribute to anxiety. In Ménière’s disease, as severe vertigo attacks are experienced which are Anxiety in Ménière’s disease 5 unpleasant and frightening, and result in a sense of loss of control and helplessness. As noted above, milder symptoms of residual or movement-provoked dizziness can also be experienced between attacks. When people with Ménière’s disease experience any dizziness, they may interpret this catastrophically, misinterpreting the symptoms as the beginning of a severe attack. Dizziness may also lead to fear that they will be in physical danger (as attacks carry a risk of injury from stumbling or falling), or a fear of embarrassment about having an attack in public or letting people down [24;25]. Negative beliefs about the consequences of vertigo have been shown to be more disabling than the symptoms themselves, leading to high levels of disability and handicap [9;26]. If people with Ménière’s disease believe that movement-provoked dizziness may develop into a severe attack, they may also believe that movement is therefore bad for them and should be avoided. This belief that physical activity may be harmful and subsequent avoidance of physical activity has also been reported among people with other chronic symptoms [16;27]. Thirdly, intolerance of uncertainty has been found to be relevant to anxiety. Many chronic illnesses result in increased levels of uncertainty with regard to the occurrence or severity of symptoms, prognosis, or the effectiveness of treatment. Uncertainty has been well noted anecdotally in Ménière’s disease and chronic vertigo [13;28;29], as attacks can occur unexpectedly, impacting on every area of life. Individual differences may occur in how people tolerate these uncertainties and adapt their lives to accept and incorporate their presence and consequences. Dugas and colleagues [30] describe someone who is intolerant of uncertainty as having “an excessive tendency to find uncertain situations stressful and upsetting, to believe that unexpected events are negative and should be avoided, and to think that being uncertain about the future is unfair” (p. 58). Intolerance of uncertainty has been Anxiety in Ménière’s disease 6 reported to lead to inaccurate appraisals of threat [30;31] and result in a greater use of vigilance and avoidance behaviours [32]. If people with Ménière’s disease believe that the unpredictable nature of their illness is stressful, unfair, and reflects badly on their character (e.g. making them appear to be disorganised or to under-perform), they may respond anxiously to all uncertain situations. They may also try to avoid situations in which unexpected attacks may occur. This may also contribute to anxiety as, due to the nature of the disease, any situation could potentially be appraised as uncertain. The purpose of this study was firstly to investigate whether illness perceptions, dizziness related fears and beliefs and intolerance of uncertainty are associated with clinical levels of anxiety, and secondly, to identify what combination of cognitions predict the maintenance of anxiety over time. Our multivariate analyses were designed to examine and control for the effects of symptom severity, in order to isolate the additional effects of these cognitions. This study was nested within a randomised controlled trial (RCT) of vestibular rehabilitation (VR) or symptom control (SC) therapy presented in the form of selfmanagement booklets for people with Ménière’s disease [33]. VR involves stimulating the balance system using a series of head movements, causing movementprovoked dizziness. The balance system gradually habituates to these movements, leading to a gradual reduction in provoked dizziness [2]. SC therapy involves the use of applied relaxation, controlled breathing and stress management strategies; the rationale is that since arousal and stress may aggravate symptoms of dizziness, reducing stress can improve adjustment and relieve symptoms [3]. By combining an observational study with this RCT we were able to examine whether the longitudinal Anxiety in Ménière’s disease 7 predictors of anxiety differed in those undertaking different self-management programmes. It was hypothesised that in line with previous research on illness perceptions, anxiety would be associated with the belief that the illness has serious consequences, belief in a chronic timeline, low perceived control, less understanding of the illness and greater emotional response. Greater levels of anxiety were also hypothesised to be associated with negative beliefs about dizziness, and a greater intolerance of uncertainty. Finally, it was hypothesised that these associations would be moderated by intervention group. As the VR intervention requires the deliberate provocation of unpleasant symptoms, stronger associations were hypothesised to occur within the VR intervention group than the SC or control groups. Method Participants and Procedure Participants were 358 members of the Ménière’s Society with current dizziness symptoms but reporting no acute attack in the previous six weeks. They were randomised to receive a self-management booklet on vestibular rehabilitation (VR) or symptom control (SC), or were assigned to a waiting list control group [33]. Questionnaire measures for this study were sent with the baseline and 3 month followup measures for the RCT. Measures Anxiety. Anxiety was assessed by the anxiety subscale of the Hospital Anxiety and Depression Scale (HADS) [34]. The HADS was chosen because it does not include somatic Anxiety in Ménière’s disease 8 symptoms of anxiety that are analogous with secondary symptoms of dizziness. Anxiety scores at baseline and follow-up were dichotomised for analysis, with participants being classified as having clinical levels of anxiety if they scored eight or more [35]. Illness perceptions. Illness perceptions were measured by eight of the nine subscales of the Revised Illness Perception Questionnaire (IPQ-R) [18]. The ‘timeline acute/chronic’ subscale assesses how long the respondent expects the illness to last, and the ‘timeline cyclical’ subscale asks respondents if the illness fluctuates or is unpredictable. The ‘consequences’ subscale measures respondents’ expectations of the effects of the illness. The ‘personal control’ subscale measures respondents’ belief in personal control over the illness, whereas the ‘treatment control’ subscale measures belief in the effectiveness of treatments. The ‘illness coherence’ subscale assesses the extent to which respondents believe they understand their illness. The ‘emotional representations’ subscale measures the presence of emotional responses to the illness (e.g. depression, anger, worry, anxiety and fear). The ‘causal’ dimension asks respondents what may have caused their illness. Factor analysis (principal component analysis with varimax rotation) was used to identify any meaningful clusters of perceived causes that could be used as causal beliefs subscales. Only one clear factor emerged, which related to the belief that Ménière’s disease was caused by psychological state (e.g. stress, worry or personality), and corresponded to the ‘psychological attributions’ factor identified by Moss-Morris and colleagues [18]. Items loading over 0.5 on this factor were summed to create a subscale with good internal consistency (Cronbach’s alpha = 0.84). Anxiety in Ménière’s disease 9 Beliefs about dizziness. Three of the four subscales of the Dizziness Beliefs Scale [25] were used to measure the extent to which participants believed that dizziness would result in negative consequences. The ‘physical danger’ subscale assesses the belief that dizziness will result in being physically harmed. The ‘social incompetence’ subscale measures beliefs about the social embarrassment of becoming dizzy in public and being unable to behave normally. The ‘severe attack’ subscale measures concern that dizziness will develop into a severe attack of vertigo. The ‘serious illness’ subscale, which measures the belief that the dizziness is a sign of an underlying disease, was not used in this study because participants knew that Ménière’s disease was the cause of their dizziness. The extent to which participants believed that their symptoms could be made worse by physical activity was measured using the ‘physical activity’ subscale of the Fear Avoidance Beliefs Questionnaire (FABQ) [27]. The FABQ was originally designed for people with low back pain, and so the ‘physical activity’ subscale was adapted for the purposes of this study by replacing references to the word ‘pain’ with the word ‘vertigo’, and removing references to participants’ backs. The internal reliability for the adapted scale was acceptable (α = .79). Intolerance of uncertainty. Intolerance of uncertainty was measured using the Intolerance of Uncertainty Scale (IUS) [31]. The IUS assesses the emotional and behavioural consequences of uncertainty for respondents, their expectations that future events should be predictable and attempts to control future events. Anxiety in Ménière’s disease 10 Demographic and illness characteristics. Single items were used to assess length of time (in months) since symptoms began, gender, and age. Vertigo was assessed using the long version of the Vertigo Symptom Scale (VSS) [6]. The ‘vertigo severity’ subscale measures the frequency and severity of symptoms of vestibular origin, such as vertigo, dizziness, and imbalance. The ‘autonomic/somatic symptoms’ subscale measures autonomic symptoms that are secondary to vestibular dysfunction and symptoms of somatic anxiety and anxiety arousal. Hearing loss was assessed using five questions from the Hearing Disability Questionnaire [36] that assessed subjective severity of hearing impairment. Tinnitus and fullness in the ear were assessed using the Tinnitus Severity Index and Aural Pressure Index [37;38]. Statistical Analyses Initially, analysis of variance (ANOVA) was used to identify variables related to anxiety (non-clinical vs. clinical) at baseline. Baseline anxiety was entered into the analysis as a fixed factor, with each of the baseline variables being entered in turn as the dependent variable. We then used ANOVA to determine whether the same baseline variables predicted anxiety at follow-up, and whether intervention group (VR vs. SC vs. control group) affected this relationship. For these analyses the ANOVAs were repeated but baseline anxiety was replaced by anxiety at follow-up and treatment group was added as a second fixed factor. No interactions were found in these analyses, indicating that intervention group did not influence the relationship between baseline variables and anxiety at follow-up, and therefore data for the intervention groups were pooled for our final analyses. Anxiety in Ménière’s disease 11 These initial analyses were intended to minimise Type II error (overlooking variables related to anxiety), and so our focus was principally on the effect sizes of each variable, rather than their statistical significance. To determine which variables were associated with anxiety while controlling for Type 1 error (i.e. minimising the likelihood that relationships were identified as significant by chance), all baseline variables identified in the ANOVAs (shown in Table 1) as potentially significantly related to anxiety were entered into two hierarchical logistic regressions with anxiety at baseline and follow-up as the dependent variables. The logistic regression for anxiety at follow-up controlled for baseline levels of anxiety by entering baseline anxiety on the first step of the regression, thus allowing us to identify predictors of change in anxiety from baseline [39]. In both regressions, demographic and illness characteristics were entered together as covariates, to control for the effects of these variables. The cognitions were lastly entered together on the final step. All statistical analyses were carried out using the Statistical Package for the Social Sciences (SPSS), version 14.0 for windows. Results Participant Characteristics Of the 358 participants, 246 were female (68.7%) and 112 were male (31.3%). The age range was 28-90 years. The length of time since their symptoms began ranged from 18 to 660 months. Ten participants dropped out before the follow-up assessment (five from the VR group, four from the SC group and one from the control group), leaving 114 participants in the VR group, 115 in the SC group, and 119 in the control group. Anxiety in Ménière’s disease 12 Following the clinical cut off points recommended for the HADS [34], at baseline 56.2% of participants had at least mild clinical levels of anxiety , and 27.4% met the criteria for moderate to severe clinical levels of anxiety. At 3 month followup, 48.1% had at least mild clinical levels of anxiety, and 24.9% had moderate to severe clinical levels of anxiety. Bivariate Analyses Bivariate analyses of the associations between baseline variables and anxiety at baseline and follow-up are reported in Table 1. None of the baseline variables had different patterns of association with anxiety at follow-up in the three intervention groups (i.e. there were no significant interactions with intervention group), and so pooled analyses for the whole sample are presented. In general, a similar pattern of associations was found with anxiety at baseline and follow-up. Of the illness characteristics, higher levels of autonomic/somatic symptoms were strongly associated with clinical levels of anxiety, and anxiety was also higher among those who reported worse symptoms of vertigo, fullness in the ear and hearing disability. Among the cognitions, the variables most strongly associated with anxiety were intolerance of uncertainty and emotional responses to the illness. Participants who were clinically anxious also had stronger beliefs that dizziness could result in them losing control and being physically harmed, embarrassed or unable to fulfil social roles. Having a poor understanding of the illness and believing that it had more severe consequences were moderately associated with higher levels of anxiety. Small to moderate associations were found between anxiety and the belief that the illness was caused by psychological factors and that dizziness would develop into a severe attack of vertigo. The beliefs that treatment would not be effective in controlling their Anxiety in Ménière’s disease 13 illness, and that physical activity could make symptoms worse also had small to moderate associations with anxiety. Predictors of Anxiety at Baseline and Follow-up The results of the logistic regression indicated that 8 of the 14 variables that had been identified by ANOVA as related to anxiety independently contributed to the regression equation predicting baseline levels of clinical anxiety (see Table 2). Clinical levels of baseline anxiety were most strongly associated with reporting greater autonomic/somatic symptoms; severity of vertigo, fullness in the ear and hearing disability were no longer related to anxiety after controlling for autonomic/somatic symptoms. However, after controlling for illness severity, baseline anxiety was strongly related to being more intolerant of uncertainty, and having a greater emotional response to illness. Clinical levels of anxiety were also associated with having stronger beliefs that the illness was caused by psychological factors and that dizziness could be made worse by physical activity and would develop into a severe attack of vertigo. Higher levels of anxiety were also related to beliefs that their illness had greater consequences and that treatment would not be effective in controlling their illness. After controlling for baseline anxiety, anxiety at follow-up was no longer related to severity of vertigo, hearing loss or tinnitus, and was also no longer related to many of the baseline psychological measures that were significant in the bivariate correlations (i.e. perceived consequences, treatment control, emotional representations, psychological attributions, fear-avoidance and the belief that symptoms might herald a severe attack). However, maintenance of anxiety was predicted by three baseline variables (see Table 2). These were autonomic/somatic Anxiety in Ménière’s disease 14 symptoms, a greater intolerance of uncertainty, and reporting less understanding of their illness. Discussion The purpose of this study was to identify cognitions associated with anxiety, while examining and controlling for the effects of symptom severity, in order to isolate the additional effects of cognitions. At baseline, anxiety was related to the severity of all symptoms of Ménière’s disease except for tinnitus, but was most closely related to autonomic/somatic symptoms. The strength of this correlation is undoubtedly due in part to the fact that autonomic and somatic symptoms are an intrinsic part of anxiety. However, they can also be provoked by vestibular disorder. It seems likely that in this study autonomic/somatic symptoms were partly related to the severity of Ménière’s disease, since the other symptoms of Ménière’s disease were no longer related to anxiety after controlling for severity of autonomic/somatic symptoms. After controlling for symptom severity, most of the hypothesised relationships between anxiety and cognitions were confirmed in the cross-sectional analyses. Anxiety was associated with the belief that the illness has serious consequences, negative beliefs about the consequences of dizziness, perceived lack of understanding of the illness, a stronger emotional response to it and a greater intolerance of uncertainty. The causal direction of associations cannot be determined from crosssectional correlations; consequently, it is not possible to be certain whether high anxiety levels caused or resulted from these beliefs and attitudes. After controlling for baseline anxiety, three baseline variables predicted the maintenance of anxiety at follow-up, although it should be noted that the size of these effects was small. Since the variance these variables shared with anxiety at baseline Anxiety in Ménière’s disease 15 was partialled out, greater significance can be attached to their potential causal role in maintaining anxiety. Moreover, these longitudinal relationships were found in all three intervention groups, and the strength of the relationships was not affected by the interventions. After baseline anxiety, the next strongest predictor of persisting anxiety at follow-up was autonomic/somatic symptoms. Given the conceptual overlap between autonomic/somatic symptoms and anxiety (as measured by the HADS), it is not surprising that they were strongly associated. Nevertheless, it was essential to include autonomic/somatic symptoms in order to control for illness severity when measuring the effects of the psychological variables. Of more interest, therefore, is the finding that intolerance of uncertainty predicted persisting anxiety after controlling for baseline anxiety and autonomic/somatic symptoms. This provides for the first time an indication that the strong association with anxiety observed in the cross-sectional analyses at baseline (and in another study [40]) may reflect a causal relationship, whereby the predisposition to react negatively to uncertainty may contribute to anxiety. In addition, a perceived lack of comprehension of the illness at baseline predicted persisting anxiety, suggesting that anxiety is maintained not only by the sense that symptoms are unpredictable but also by the sense that they are inexplicable. The findings of this study cannot be generalised to all people with Ménière’s disease, as the RCT was limited to participants from the Ménière’s Society who had current dizziness but were not experiencing frequent spontaneous attacks of acute vertigo. Members of the Ménière’s society may not be representative of the general medical population of people with Ménière’s disease. For example, members may have wanted to join the society as a result of higher levels of anxiety than non members. Therefore, these findings need to be replicated in a sample who have not Anxiety in Ménière’s disease 16 joined a self-help group. A further limitation of this study is that we were only able to analyse and report associations with one aspect of the distress caused by Ménière’s disease. It is probable that other key aspects of distress, such as depression and handicap, are related to different patterns of symptoms and cognitions [40]. Most importantly, while longitudinal prediction of changes in anxiety provides stronger evidence of possible causality than can be inferred from cross-sectional associations, it cannot confirm a causal relationship. In order to do this it would be necessary to show that the outcome of an intervention that was successful in reducing anxiety was mediated by a reduction in autonomic/somatic symptoms and intolerance of uncertainty. About half of the participants in our RCT had possible clinical levels of anxiety at baseline. This observation is consistent with the findings of Savastano and colleagues [41], who identified distressed and non-distressed subgroups, and suggests that whereas some people with Ménière’s disease are able to successfully adjust to having the disease, others may need support to achieve this. In the RCT in which this study was embedded [33], vestibular rehabilitation (VR) resulted in a reduction in symptoms (assessed by a scale that measured both vertigo and autonomic/somatic symptoms) and a decrease in anxiety (measured by the HADS). Since VR requires patients to deliberately and repeatedly provoke dizziness (in order to stimulate neurological adaptation), undertaking VR teaches patients that residual symptoms are tolerable, and partly predictable and controllable, and that it is not necessary to avoid activity. It has therefore been suggested that VR can function as a form of cognitivebehavioural therapy, interrupting the vicious cycle of symptoms and anxiety about symptoms [3;33;42]. Indeed, in the trial associated with this study, VR also resulted in a reduction in negative beliefs about dizziness. Nevertheless, benefits obtained Anxiety in Ménière’s disease 17 using the self-help booklets were modest, and further research is needed to identify additional therapy components that might improve outcomes. The cognitions found to be relevant to anxiety in this study are consistent with results found among populations with chronic pain [22], multiple sclerosis [23] and dizziness [25;43], and suggest that there may be a pattern in how people perceive and think about chronic illness that is related to poor adjustment and increased distress. Emotions, bodily symptoms, cognitions and behaviours seem to become linked in a strong and unhelpful way, and it may be necessary for cognitive-behavioural therapy to address all of these components of the illness experience. However, this study has identified intolerance of uncertainty as a reaction that may be particularly important to address in therapy for Ménière’s disease. Action and contingency plans might be useful coping tools. However, McCracken and Eccleston [44] suggest that interventions that focus on acceptance rather than coping with chronic illness may be more beneficial in improving adjustment. As uncertainty cannot be avoided in Ménière’s disease, support could be focused on helping people with Ménière’s disease to accept that at times they may not be able to do certain things. A controlled trial is needed to test whether anxiety can be reduced by successfully treating intolerance of uncertainty. Acknowledgments This study was funded by the Ménière’s Society, UK. 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