Patient perceptions of the burden of coeliac disease and its treatment in the UK
Prof. R. F. A. Logan, Division of Epidemiology and Public Health, School of Community Health Sciences, University of Nottingham Medical School, Nottingham, NG7 2UH, UK.
Background Coeliac disease affects about 1% of the population, with the majority being undetected. As a consequence, there have been calls for the introduction of screening. Before screening is given serious consideration, it is important to assess how acceptable early diagnoses and treatment would be.
Aim To assess patients’ views as to the diagnosis and treatment of disease.
Methods Coeliac disease patients who had taken a gluten-free diet for at least 12 months (mean 60 months) were mailed a questionnaire. Coeliac patients presenting with typical classical symptoms were compared with those diagnosed without such symptoms.
Results Overall, 83% (147/177) of coeliac patients returned the questionnaires. Two-thirds (68%, 101/147) reported that their dietary restrictions reduced their enjoyment of food; 46% (68/147) believed their food cost them more and estimated this to be an extra £10 (€16) per week. Of those reporting greater cost, 31 (21%) said this was a problem for them. Half (54%, 80/147) reported doing things they enjoyed less often because of their diet, with the most common activity sacrificed being dining out (n = 65). In spite of these findings, 81% (119/147) reported being pleased that they were diagnosed, with 66% (59/89) of cases with classical symptoms wishing they had been diagnosed earlier compared with 45% (23/51) of those without such symptoms (χ2 = 6.0, P < .05). In contrast, 27% (14/51) of coeliacs diagnosed without classical symptoms regretted being diagnosed with their condition compared with 10% (9/89) of those with classical symptoms (χ2 = 7.1, P < .01).
Conclusions Even after several years of a gluten-free diet, many patients with coeliac disease regard it as a substantial burden, with a quarter of screen detected patients reporting regret at being diagnosed. Our findings question how acceptable screening for coeliac disease would be in people with minimal or no symptoms.
Recent population studies have shown that coeliac disease affects about 1% of the general population in many countries and a vast majority are undiagnosed.1–4 There have, therefore, been calls to establish population-based screening programmes to diagnose and treat the undiagnosed population.5,6 However, the extent of any morbidity associated with undiagnosed coeliac disease is unclear.2,7,8 In addition, it is uncertain whether people with asymptomatic coeliac disease will accept the burden of life long adherence to a gluten-free diet.
The aim of this study was to assess how patients with coeliac disease currently perceive the burden of their disease and its treatment and to compare any burden between coeliac patients who presented with classical symptoms of the disease such as diarrhoea which also would be expected to improve on a gluten-free diet (GFD) and those patients who presented for diagnosis without reporting such symptoms of the disease.
Patients with coeliac disease were identified in the Nottingham and Derby area of the UK. All patients approached had a biopsy confirmed diagnosis of coeliac disease, had been prescribed a gluten-free diet and had been given advice about their diet from a dietician. The patients from Nottingham were identified from the records maintained by the dieticians at the Queens Medical Centre, Nottingham. The patients from Derby were identified from a comprehensive database of all known coeliac patients attending either the Derby Royal Infirmary or Derby City Hospital maintained by one of the authors (GKTH). To ensure that some patients had taken a GFD for several years the patients taken from the database were those who had presented for diagnosis in the years 1996–7 and 2001, 5 years later.
All patients were between the ages of 18 and 75, had the diagnosis confirmed by duodenal biopsy and had been on a gluten-free diet for at least 12 months according to their records. This interval was chosen to allow sufficient time for patients to have adjusted to the new diet and to allow any symptoms related to coeliac disease to resolve on a strict gluten-free diet. Hospital records were obtained for 177 coeliacs (Derby 104, Nottingham 73). The patients then were divided into patients who had presented with typical classical symptoms of the disease, namely any abdominal symptoms, weight loss, lethargy, tiredness, and those who presented with other symptoms for which there was a wide range of reasons for presentation (musculo-skeletal complaints-18, mild and asymptomatic anaemia-6, family history-5, osteoporosis-2, screening of associated conditions-8, miscellaneous-13). Insufficient information was available to classify seven patients.
A questionnaire was designed adapting questions used by other authors as well as creating novel questions from the clinical experience of both gastroenterologists and dieticians9 (Appendix S1). The questionnaire was piloted by interview with 10 patients. Following piloting, the wording of the questions were confirmed as comprehensible and further questions were developed as to ensure that the questionnaire fully addressed the impact of a diagnosis of coeliac disease and of having to take a gluten-free diet. The questionnaire was then mailed, with a reminder being sent to initial nonresponders after 6 weeks. Data were entered and subsequently analysed using spss version 11.0 (SPSS, Chicago, IL, USA). Chi-square tests were used to examine differences between groups.
Ethical Committee approval was obtained from Nottingham Health Authority LREC and Southern Derbyshire LREC.
Of the 177 coeliac patients approached, 147 returned questionnaires (response rate 83%). 25 (14%) patients failed to respond, one patient had died and four patients denied ever having been recommended a gluten-free diet and so were not included in analysis. Of the 62 patients who presented without classical symptoms of the disease, 51 (82%) responded. Nine (15%) failed to respond and 2 (3%) denied being on a gluten-free diet. Of the 108 patients presenting with classical symptoms of coeliac disease, 89 (82%) responded. 17 (15%) failed to respond and 2 (2%) denied being on a gluten-free diet and so were not included in the analysis. The characteristics of the coeliac patients who responded are shown in Table 1; there was no significant difference in age or length of time on diet across the type of presentation groups.
Table 1. Characteristics of coeliac disease patients
|Responders||147/177 (83%)||89/108 (82%)||51/62 (82%)|
|Median age (years) (IQ range)||54 (44–62)||54 (45–63)||52 (39–62)|
|Gender||M = 38 (26%)||M = 21 (24%)||M = 15 (29%)|
|F = 109 (74%)||F = 68 (76%)||F = 36 (71%)|
|Length of time on diet in months median (IQ range)||45 (29–76)||48 (30–78)||35 (28–73)|
|When you were diagnosed your health was as far as you can remember|
| Excellent||6 (4%) ||2 (2%)||3 (6%)|
| Very good||13 (9%)||4 (5%)||8 (16%)|
| Good||25 (17%)||15 (17%)||10 (20%)|
| Fair||38 (26%)||24 (27%)||13 (26%)|
| Poor||39 (27%)||23 (26%)||12 (24%)|
| Very poor||26 (18%)||21 (24%)||5 (10%)|
As shown in Table 2, 68% (101/147) of all coeliac patients reported that their dietary restrictions reduced their enjoyment of food. 46% (68/147) believed their food cost them more than people without dietary restrictions and perceived this to be about £10 (€15) per week. Of those reporting greater cost of food, 31 (21%) said this was a problem for them.
Table 2. Reported compliance with gluten-free diet
|How often do you eat food items that you are advised to avoid?|
| Every day or most days||7 (5)||2 (2)||5 (1)|
| Once a month or more but not most days||32 (22)||18 (20)||12 (22)|
| Less than once a month or never||98 (67)||63 (71)||31 (61)|
| Missing||10 (7)||6 (7)||3 (6)|
|Does your dietary restriction affect enjoyment of food?|
| Greatly increased||5 (3)||4 (5)||0|
| Slightly increased||4 (3)||2 (2)||2 (4)|
| No effect||28 (19)||14 (16)||12 (24)|
| Slightly reduced ||58 (40)||38 (43)||18 (35)|
| Greatly reduced||43 (29)||26 (30)||16 (31)|
| Missing||9 (6)||5 (6)||3 (6)|
About half of all coeliacs (54%, 80/147) reported doing things they enjoyed less often because of their dietary restriction (Table 3). From the free text responses, the most common activity sacrificed was reported as eating out (n = 65). As Table 4 shows 52% of coeliac patients (76/147) felt as though they were different from other people, 65% (96/147) reported frequent feelings of frustration and 56% (83/147) of coeliacs reported being more worried about their health because of their diagnosis.
Table 3. Social burden
|Are there things that you enjoy doing that you now do less often because you are on a specially prescribed diet?|
| Yes||80 (54)||55 (62)||20 (39)|
| No||66 (45)||33 (37)||31 (61)|
| Missing||1 (1)||1 (1)||0|
|Did your socializing with friends at work and during leisure hours change when you started on the diet?|
| I socialized a lot more||1 (1)||1 (1)||0|
| I socialized a little more||4 (3)||4 (4)||0|
| Socializing didn’t change||78 (53)||40 (45)||33 (65)|
| I socialized a little less||33 (22)||23 (26)||10 (20)|
| I socialized a lot less||21 (14)||15 (17)||5 (10)|
| Missing||10 (7)||6 (7)||3 (6)|
Table 4. Psychological burden
|Because you have been diagnosed with the condition that you are taking a prescribed diet for, does it make you feel as though you are different from other people?|
| No||70 (48)||42 (47)||23 (45)|
| Yes||76 (52)||46 (52)||28 (55)|
| Missing||1 (1)||1 (1)||0|
|Do you ever feel frustrated because of the special diet you have been told to stick to?|
| Every day||24 (16)||18 (20)||6 (12)|
| At least once a week||39 (27)||26 (29)||11 (22)|
| At least once a month||33 (22)||18 (20)||15 (29)|
| Less than once a month or never||50 (34)||27 (30)||19 (37)|
| Missing||1 (1)||0||0|
|Because you have been diagnosed with the condition that you’re on the prescribed diet for, do you think that you worry about your health more than most people?|
| Yes I worry a lot more||19 (13)||15 (17)||4 (8)|
| Yes, I worry a bit more||64 (44)||36 (40)||24 (47)|
| No, I worry a bit less||34 (23)||19 (21)||13 (26)|
| No, I worry a lot less||20 (14)||11 (12)||8 (16)|
| Missing||10 (7)||8 (9)||2 (4)|
In spite of this, 81% (119/147) reported being pleased that they were diagnosed with 66% (59/89) of cases with classical symptoms wishing they had been diagnosed earlier compared with 45% (23/51) of those without these symptoms (χ2 = 6.0, P < 0.05) (Table 5). In contrast, 27% (14/51) of coeliac patients diagnosed without classical symptoms of the disease regretted being diagnosed with their condition compared with 10% (9/89) of those with classical symptoms (χ2 = 7.1, P < 0.05).
Table 5. Satisfaction of diagnosis
|Do you believe that your life would have been better if you had been diagnosed earlier?|
| No, much worse||7 (5)||5 (6)||2 (4)|
| No slightly worse||6 (4)||1 (1)||5 (10)|
| Much the same||46 (31)||23 (26)||21 (41)|
| Yes, slightly better||44 (30)||29 (33)||15 (29)|
| Yes, much better||43 (29)||30 (34)||8 (16)|
| Missing||1 (1)||1 (1)||0|
|Are you pleased that you have been diagnosed when you consider life before and after diagnosis, bearing in mind the changes to your life that you’ve had to make?|
| Yes, very pleased||77 (52)||47 (52)||25 (49)|
| Yes, somewhat pleased||42 (29)||30 (34)||12 (24)|
| Not particularly pleased||19 (13)||7 (8)||10 (20)|
| Not at all pleased||6 (4)||2 (2)||4 (8)|
| Missing||3 (2)||2 (2)||0|
Our study found that for most patients with coeliac disease, the condition was continuing to have a substantial impact on their lives several years after diagnosis. Over two-thirds reported reduced enjoyment of food, 36% reported less social activity, 43% regular frustration with the dietary restriction and 57% greater anxiety about their health. In addition, food was costlier and a half felt stigmatized. As a whole, these negative responses were reported as often in those with minimal or no symptoms at diagnosis as in the group with classical symptoms. Thus, even after several years, many patients clearly regard their coeliac disease as intrusive and burdensome.
In spite of these findings, over 80% reported being pleased at having been diagnosed with coeliac disease and almost a half of those in the minimal or no symptom group believed that their lives would have been better with earlier diagnosis. To what extent this reflects a real improvement in health or simply a perceived health benefit in terms of a possibly reduced risk of coeliac disease complications is not possible to assess from our study. Nevertheless, the greatest difference between the two groups was in regret at being diagnosed with 27% of the minimal or no symptom group expressing regret compared with 10% of the classical symptom group.
How valid are these findings? Patient responses to interviews or questionnaires can readily be influenced by context. One advantage to using a postal questionnaire is absence of interviewer introduced bias. We took care to ask the questions in a neutral manner as can be seen from their wording as shown in the questionnaire in Appendix S1. In addition, the senders of the questionnaire were not known to or directly involved in care of these patients. The response rate was excellent and similar in the two groups. Inevitably, when using a 5-point Likert scale, response numbers in some groups were small necessitating combining responses. A larger study might have allowed us to tease out whether some of the extreme responses differed between those with classical symptoms and the minimal or no symptom group.
Previous studies have explored aspects of disease burden associated with a diagnosis of coeliac disease, particularly in relation to the psychological, sociological and economic burden.10–12 However, this study is, to our knowledge, the first to do so in the UK. Other researchers have also previously compared populations of coeliac patients presenting both with and without symptoms. These have focused on aspects such as quality of life scores and gluten-free dietary compliance.13,14 This study goes a step further in attempting to compare patients’ specific perceptions of the burden of their condition between the two different patient groups. It also explores the question of regret towards receiving a diagnosis, clearly of importance when considering any potential screening programme.
Our findings are in keeping with some other recent studies, which have focused on quality of life and the affective state of patients taking a gluten-free diet. Hallert and colleagues in Sweden were the first to highlight the fact that adults with coeliac disease, despite having what seemed an easily and effectively treated condition, reported poor subjective health on the SF36 questionnaire and were often depressed.9,15 Similar findings have been reported from studies in Italy and Germany.16–18 Most recently, Addolorato et al. have found a 70% prevalence of social anxiety disorder (also called social phobia) in coeliac disease patients compared with only 16% in their healthy controls and the presence of social phobia correlated with being depressed.19,20
In contrast, studies from Finland and North America have reported fewer problems in coeliac patients established on their gluten-free diet.13,21–23 In particular, Viljamaa et al. used interviews and questionnaires to assess compliance, quality of life and psychological well-being in 53 screen-detected coeliacs and 44 symptom-detected coeliacs and compared them with population controls.23 No differences in dietary compliance and quality of life were seen between the screen-detected and symptomatic groups. Psychological well-being scores and quality of life using SF36 was also similar to that in the population controls. The authors speculate that their good results may reflect greater awareness of coeliac disease in a country where the prevalence of diagnosed coeliac disease is high. Another factor may be that in these studies, many of the screen-detected cases were diagnosed from screening family members. Such cases will be self-selected from family members already aware and willing to accept the constraints of a gluten-free diet; or put alternatively, family members not willing to restrict their diet will tend not to accept or seek coeliac disease screening.
Larger studies are clearly needed. In the meantime, our findings suggest that if screening was to be introduced in the UK, at least a gluten-free diet would be unacceptable to a significant minority of screen-detected cases.
Declaration of personal and funding interests: None.