Annotated Bibliography: Gluten Free Diet Adherence

Leffler, D., Edwards-George, J., Dennis, M., Schuppan, D., Cook, F., Franko, D., Blom-Hoffman, J., & Kelly, C. (2007). Factors that Influence Adherence to a Gluten-Free Diet in Adults with Celiac Disease. Digestive Diseases and Sciences, 53, 1573-1581.

The purpose of this study was to determine factors that contribute to GFD adherence in a population of adults with CD in the New England area. A questionnaire was developed employing an expert panel, which included gastroenterologists, nutritionists, psychologists, and adults diagnosed with CD. Through this process a set of domains relevant to GFD adherence were identified (i.e., the psychosocial burden of disease, symptoms, social and health support, self-efficacy, perceived adherence, and general health), and the resulting questions were tested on two focus groups of 8-12 adults with biopsy confirmed CD. The final questionnaire, named the Global Celiac Assessment Scale (GCAS), contained 142 items. Adults (≥ 18 years old) who had been diagnosed with biopsy-confirmed CD for longer than three months were recruited via posters at CD support group meetings, advertisements in CD newsletters, and from the Celiac Center at Beth Israel Deaconess Medical Center (BIDMC). Once recruited, 154 participants (77% female) completed the GCAS, and had their blood tested for IgA antibodies and their GFD adherence levels assessed by a skilled, CD-knowledgeable nutritionist. Results of this study indicated that understanding of the GFD, membership in a CD support group, and self-efficacy were the greatest factors in determining GFD adherence. Unlike other studies of this topic, gender was not a factor in determining adherence, anxiety, or self-efficacy levels. However, married participants had higher adherence levels than non-married participants. Surprisingly, patients significantly overestimated their adherence levels; 70.1% reported strict adherence to the GFD, but only 44.2% were assessed at ‘Excellent’ and 34.4% at ‘Good’ adherence levels by the nutritionist. Participants also reported less than 70% satisfaction ratings with the amount of adequate support and knowledge received from their health care providers, who included dieticians (63%), gastroenterologists (57.1%), primary care physicians (35.7%) and pharmacists (22.7%). Likewise, participants reported the Internet as the most useful source of information (85.1%), and even rated their friends without CD as better sources of information than their primary care physician (44.8% vs. 24.7%).

Possibly the most interesting finding reported in this study was the idea that gender was not a factor in GFD adherence for participants in this population, as multiple studies have shown that generally females with CD report higher rates of adherence than men. However, because the researchers recruited a small sample of participants from the New England area, it is difficult to assess whether these findings could be replicated within other parts of the United States. Generally speaking, this particular area of the country tends to have more affluent citizens and greater availability of GF resources, grocery items, and restaurants. In addition, participants were recruited through support groups, a specialized celiac health center (at BIDMC), and through CD newsletters, indicating that people with CD who were not associated with those programs and/or had not received those resources were not given an opportunity to participate in this study. Unfortunately, that factor may have contributed to selection bias. The finding that married couples had higher adherence than those who were not married was also, arguably, one of the most important pieces of information uncovered by this study. This was one of the first and only projects, to date, to have investigating marital status in relation to GFD adherence. If these findings are replicable, this could have serious implications for the development of future research and interventions aimed at understanding and increasing the psychological factors necessary for enhancing positive well-being and treatment adherence for celiac patients. Additionally, it was surprising and disturbing to find that, although 70% of participants claimed strict adherence, less than 50% actually participated in a strict GFD. Many countries have released statistics stating GFD adherence levels to be generally between 60% - 80%. Considering the findings from this study, it would be interesting to explore the actual versus perceived levels of adherence to the prescribed GFD in other areas within the United States and internationally.

Annotated Bibliography: Quality of Life for Celiacs

Ciacci, C., D’Agate, C., De Rosa, A., Franzese, C., Errichiello, S., Gasperi, V., Pardi, A., Quagliata, D., Visentini, S., & Greco, L. (2003). Self-Rated Quality of Life in Celiac Disease. Digestive Diseases and Sciences, 48 (11), 2216-2220.

This study was conducted in Italy, with the purpose of assessing the self-perceived quality of life in CD patients who had adhered to a GFD for at least one year. The Zung Self-Rating Depression Scale – in combination with questions regarding CD knowledge levels, demographics, and GFD adherence – was administered to 745 members of the Italian Celiac Society, which represented five regions of Italy. Seventy eight percent (N = 581, 71% female, M_age = 31.5) of the questionnaires were completed. A majority of the respondents (96.9%) showed a strong understanding of CD (e.g., what it was, the necessity of a GFD, and the genetic risks associated); however, only 74.1% reported strict adherence to the diet. According to the questionnaire, reasons for transgressing were: problems ordering in restaurants, feelings of anger toward CD, not to be different from others, and hope that small amounts of gluten would not be harmful. Fifteen percent of participants ‘often’ or ‘very often’ felt embarrassed by having to ask for GF food at restaurants or parties. Results also showed that females adhered to the GFD more strictly, had lower happiness scores, and had slightly higher anxiety levels than men. Patients who had been diagnosed before the age of twenty had significantly higher happiness scores than those diagnosed in adulthood, although overall sense of well-being was higher in patients diagnosed in adulthood. In this study, as in others of its kind, QOL scores did not differ greatly from the overall population. In fact, 83.6% of participants indicated that their overall feelings of well-being were ‘very well’ and ‘well’. This study also showed that the actual duration of a GFD was not correlated with any one item on the questionnaire, disproving the authors’ hypothesis that adherence time would affect QOL scores. The authors wrote that self-reported high (or unaffected) QOL scores in CD patients may have been partially influenced by the enthusiastic recovery that many experienced after initiating a GFD. However, they questioned whether the higher compliance rate of women to the GFD (as compared with men) contributed to the increased levels of anxiety and lower levels of happiness indicated in the study.

Multiple variables were collected and cross-correlated for this study, including age, gender, knowledge levels, GFD adherence, age at diagnosis, and patient emotions (anxiety, depression, and happiness). Because of this measurement depth, this study provided a broader understanding of the study sample, as well as how emotion levels varied depending on the patient’s age at the time of the study, their age at the time of diagnosis, and their gender. Few studies have analyzed data to this extent when working with a celiac community. Unfortunately, the number of emotions investigated limited this study. By only assessing anxiety, depression, happiness, and overall well-being it was difficult to determine why happiness levels were low, when well-being scores were relatively high and depression and anxiety levels were relatively low. It would have been beneficial to clarify the definition of well-being for the participants of this study, or to have included additional positive emotion levels in the questionnaire to create a more accurate depiction of the psychological processes involved in GFD adherence. In addition to depression and anxiety, this study would have benefitted from investigating shame, resilience, and coping. Future studies of this kind should employ a battery of questionnaires aimed at capturing a more comprehensive picture of the emotional and stress-related processes experienced by celiac patients. Additionally, by only choosing participants from the Italian Celiac Society, the authors risked selection biases, which may have contributed to their participants’ relatively high levels of understanding of CD, GFD adherence, and overall well-being. Individuals engaged in treatment support groups may feel higher levels of perceived support, social inclusion, and have more access to nutrition and GFD information than the general celiac population. Future studies should make more of an effort to include participants from venues other than celiac support groups and hospitals, such as individuals who have self-diagnosed as celiac and/or those in low socioeconomic areas with limited access to adequate health resources and medical specialists.

Annotated Bibliography: Gender Differences in Celiac

Sverker, A., Ostlund, G., Hallert, C., & Hensing, G. (2009). ‘I lose all these hours…’ – Exploring gender and consequences of dilemmas experienced in everyday life with coeliac disease. Scandinavian Journal of Caring Sciences, 23, 342-352.

The purpose of this article was to further explore the often referenced and ill-defined presumption that women with celiac disease (CD) experience lower levels of psychological well-being and health related quality of life (HRQoL) than men with CD. In addition to investigating this phenomenon, the authors elected to include close male and female relatives, in order to compare and contrast daily perceptions of stress associated with exposure to an individual restricted to a strict gluten-free diet (GFD). This project employed a mixed-methods approach, including both the implementation of qualitative interviews and a newly developed celiac questionnaire. The survey element was employed to capture gender norms within celiac family relationships (e.g., who primarily does the cooking, preparing, and shopping?), to address previous researchers’ hypotheses that gender norms may be the cause of greater distress experienced by women with CD. Interestingly, the authors’ found that within their sample (N = 43 CD patients, 74% female; N = 23 close relatives, 74% male) females with CD and male close relatives experienced the same stressor perceptions when it came to cost of food items, time (e.g., extra time spent preparing meals), tiresome comments from others when eating out, and difficulty traveling and/or enjoying holidays. Women with celiac and male close relatives were also less inclined to engage in meals with others, in order to avoid feelings of social stigma and ostracism. Men with celiac did not experience these extra stressors, supposedly because the shopping for and cooking of gluten-free items was primarily performed by someone other than himself. However, both men and women with CD reported feeling shame as a result of dilemmas associated with GFD adherence, and experienced similar levels of chronic anxiety associated with gluten contamination and dinner party invitations (i.e., considering the potential for gluten-free options, and declining participation).

This study was one of the first to directly address daily stressors and gender differences in coping with celiac disease. It is a highly influential piece, in that it speaks to not only the stresses and coping processes specific to females with celiac as compared to males, but also includes the perspectives of spouses and close relatives of patients. One of the most interesting factors addressed in this paper is the notion of a “triple burden” experienced by women, which is attributable to their necessity to engage in paid and unpaid work in addition to caring for a chronic illness. As a result of these extra burdens placed on women, they are prone to lose more time out of their day and make extraordinary sacrifices in order to care for themselves and their families. The implications for this hypothesis supply a wealth of material from which to conduct future research. If women do, as this and other studies describe, experience increased burden of disease and daily personal stressors, additional studies must be conducted to further investigate the frequency and effects of these events and the psychological factors surrounding interpersonal interactions and support. For instance, considering this study’s findings that male close relatives experience similar levels of dilemma associated with GFD adherence, do their associated females with celiac adequately perceive the partners’ emotional empathy and support behaviors? Do these women find their partners’ sympathy comforting, stressful, annoying, etc.? It is possible, in considering these potential research questions, that women may feel an additional psychological burden associated with comforting their partners’ distress from celiac-related dilemmas in addition to coping with their own. In this case, male relative support would be more hurtful than helpful, and should be measured and addressed in future studies. Despite the influential nature of this study, it is important to consider the fact that the authors failed to clearly define their illness population, often referring to participants as “gluten intolerant”. Gluten intolerance is not the same as celiac disease, as symptoms of gluten intolerance are unrelated to immune system functioning and neurological processes (i.e., generally related to gastrointestinal issues exclusively). From a health perspective, this is an important distinction that should have been better addressed. However, despite that slight misstep, this study provides incredibly valuable material from which to begin properly understanding and addressing the questions surrounding gender differences and social support in living with celiac disease and similar diet-related chronic illnesses.

Presentation: Celiac and the Internet

On October 29th I presented a workshop at the 2011 Annual Northwest ECO Conference, aimed toward discussing the role of the Internet in empowering and promoting change within the celiac community. Below are the slides from that presentation.

Obtaining a Celiac Diagnosis

For many of you it is no surprise that obtaining a diagnosis of celiac (or even being administered a test for it) is extremely difficult. In fact, research has shown that most people with celiac go an average of 13 years from the onset of symptoms to diagnosis and visit their physician approximately 30 times (Kostopoulou, Devereaux-Walsh, & Delaney, 2009). It would be easy to paint the doctors as the villains in these scenarios - "If they would only listen to me, they would have tested me a long time ago!" - but in reality we are victims of a disorder that has many and varying symptoms, making it extremely difficult to pinpoint. In my own experience, I saw my doctor multiple times complaining of stomach pain, headaches, chronic back pain, and bloating, which always resulted in him telling me essentially to "be a good girl and take some Advil." Then, in my frustrated mind, he would pat me on the head and scoot me out of the office. Was I angry? Yes. Should I have been? Maybe.

Here is a list of all of the currently recognized symptoms of celiac disease. I say "currently" because the list seems to grow, shrink, and/or change every time I read a new study or website.

• Abdominal cramps, gas and bloating
• Anemia
• Borborygmi (stomach rumbling)
• Coetaneous bleeding
• Depression
• Diarrhea
• Disinterest in normal activities
• Easy bruising
• Epitasis (nose bleeding)
• Failure to thrive
• Fatigue or general weakness
• Flatulence
• Fluid retention
• Foul-smelling or grayish stools
• Gastrointestinal hemorrhage
• Gastrointestinal symptoms
• Hematuria (red urine)
• Hypocalcaemia/ hypomagnesaemia
• Inability to Concentrate
• Infertility
• Iron deficiency anemia
• Irritable Mood
• Lymphocytic Gastritis
• Mood Changes
• Muscle wasting
• Muscle weakness
• Nausea
• Night Blindness
• No obvious physical symptoms (just fatigue, overall not feeling well)
• Obesity
• Osteoporosis
• Pallor (unhealthy pale appearance)
• Panic Attacks
• Peripheral neuropathy (nerve damage)
• Stunted growth in children
• Thyroid Disorders
• Type 1 Diabetes
• Vertigo
• Vitamin B12 deficiency
• Vitamin D deficiency
• Vitamin K deficiency
• Vomiting
• Voracious appetite
• Weight loss

Technically, you only need to recognize that you're experiencing at least three of these symptoms to qualify for a biopsy or blood test. However, insurance companies and physicians tend to 'play it safe' and only test once all other possibilities are excluded or the patient has been experiencing the symptoms (and complained for the tests) long enough to justify a celiac hypothesis. Again, though, this is not because they are evil people who are okay with watching their patients suffer. Look at this list again. Seriously look at it. Any combination of the symptoms from this list could easily be misinterpreted to mean a variety of illnesses, and most of the combinations (like the one I was reporting to my physician) seem like short-term, normal issues that should go away with time. Thirteen years is certainly too much time, but that's why so many of us are working hard to raise awareness for celiac. In theory, the more aware patients are going in to the doctor's office, the more likely that they will walk away with the results they need. Knowing that you suffer from some of the symptoms on this list is enough evidence to really cause a stink next time you, or someone you know, seek out a celiac test.

Just remember that even though physicians can certainly drag their feet, and the gluten-free diet can be easily self-prescribed as a treatment, you CAN NOT be accurately tested for celiac unless you are actively consuming gluten ingredients. If having a medical diagnosis is important to you (i.e., if you expect to visit the hospital in the future and want to ensure that you are given gluten-free foods and medications), do not initiate a gluten-free diet on your own.

References

Kostopoulou, O., Devereax-Walsh, C., & Delaney, B.C. (2009). Missing celiac disease in family medicine: The importance of hypothesis generation. Medical Decision Making, 29, 282-289.

Article Review: Psychological Support Counseling Improves Gluten-Free Diet Compliance in Celiac Patients With Affective Disorders

Summary

Background

Celiac disease (CD) is an autoimmune disorder, which involves the breakdown of mucosal villi in the small intestine upon exposure to gluten (i.e., wheat, barley, rye). Symptoms of CD can be neurological (e.g., depression, anxiety, ADHD, fatigue, etc.), physical (e.g., flatulence, diarrhea, bloating, rash, etc.), or a combination of the two. The only known treatment to date is life-long adherence to a strict 100% gluten-free diet (GFD). However, for many patients compliance to the prescribed diet is difficult and transgressions are common. In addition, depression and anxiety are traits commonly found in patients with CD, and are among the only symptoms of the disease to not fully dissipate after initiation of the GFD. Therefore, the authors of this study hypothesized that psychological support counseling for celiac sufferers with depressive symptoms would increase the likelihood of strict compliance to the prescribed GFD.

Methods

In order to test the effectiveness of psychological support on the target population, the authors of this study conducted a randomized experiment. Sixty six newly-diagnosed celiacs from an out-patient clinic were randomly divided into two groups based on the independent variable: psychological support. Group A received free counseling sessions every two weeks following the initial diagnosis. Group B acted as the control group and received no support. The dependent variables in this study were: 1) compliance to the GFD, 2) reduction in anxiety, and 3) reduction in depression levels. Patients in both groups were tracked for six months, and were asked for a blood sample every two months to test for AGA and EMA antibodies (indicating the presence of gluten). Adherence to the GFD was determined by AGA and EMA disappearance, self-report, family member interview, and histological recovery.

Results

At the end of six months, fewer patients in group A displayed state anxiety and current depression characteristics as compared to patients in group B (5 vs. 26). Only three patients in group A had poor compliance to the GFD, as opposed to 13 patients in group B. Non-compliance to the GFD was significantly higher for those who were not provided psychological support.

Discussion

The authors recognized that this is the first study to collect data on the benefits of providing psychological support to newly-diagnosed celiac patients. As predicted, participation in regularly scheduled counseling sessions reduced the percentage of depressed CD patients and increased the likelihood of strict diet adherence. To this end, they proposed that in a cost/benefit analysis it is less expensive for the patient to receive regular counseling than it is to incur the long-term costs associated with poor adherence to the GFD (e.g., hospital bills, vitamins, over-the-counter drugs, etc.). The authors also suggested that this method would benefit people suffering from other chronic diseases, such as diabetes.

Further Analysis

Strengths of the Study

This study is a breakthrough in understanding treatment and dietary promotion techniques within the celiac community. It is one of a very few number of experiments conducted within this population (most psychology-related CD research is based in survey, interview, and/or focus group processes), and it is the first experimental study to test the benefits of support for GFD adherence. In addition, the retention rate of the study was 100%, indicating the level of importance and trust CD patients (especially newly-diagnosed ones) place on the doctor/patient relationship.

Weaknesses of the Study

Although this study is revolutionary in the field, it fails to take into account a few important factors which are commonly left out of celiac research. The role of family support, the number of people in the household also diagnosed with CD, and the ease of access to GF foods and information are not mentioned in the article. It can be inferred, however, that patients enrolled in the study were receiving at least some family support with regards to the GFD, because a family member interview was used as one of the determinants of GFD adherence at the end of the study.

Future Research

This study is an important jumping off point for understanding the connections between psychological symptoms associated with CD and success in GFD adherence, but the results fail to explore the more personal connections surrounding celiac patients. Although multiple studies of diabetes, cancer, and Crohn’s disease explore the benefits of marital and family support, this topic has yet to be explored for CD. However, with an estimated 1 in 133 Americans living with this disorder future research must be conducted to determine the causal relationship between family support, informational support, and GFD adherence.

References

Addolorato, G., De Lorenzi, G., Abenavoli, L., Leggio, L., Capristo, E., & Gasbarrini, G. (2004). Psychological support counseling improves gluten-free diet compliance in celiac patients with affective disorders. Alimentary Pharmacology & Therapeutics, 20, 777-782.