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A case of functional symptoms in treated coeliac disease ?

Nick Trott RD. Specialist Gastroenterology Dietitian, Royal Hallamshire Hospital, Sheffield

Mrs X was a married 35 year old with two young children. She was self-employed and her job entailed extensive travel. Prior to her referral into our coeliac and gluten sensitivity service she had presented to her GP complaining of fatigue and gastrointestinal symptoms, specifically diarrhoea and chronic abdominal pain. 
Mrs X was referred to a consultant gastroenterologist within our hospital who confirmed a diagnosis of coeliac disease (CD) on the basis of positive corroborative serology and duodenal biopsy (see Table 1).

 

Table 1

Serology Baseline Level 4 month Follow up Ref Range
tTG 77 3 0-7 U/ml
EMA Strong Positive Negative N/A
Histopathology Marsh Grade 3c Marsh Grade 1 N/A
Ferritin 35 78 Ug/L
Folate 4 12.7 >4.6 ug/L
B12 246 653 197-771 ng/L
Vitamin D 26 54 Nmol/L >30 (> 50 ideal)
DEXA Bone Scan Osteopenic N/A N/A

Mrs X was referred into dietetics for further advice and support in relation to establishing a strict gluten-free diet (GFD). Over the next two years Mrs X was regularly reviewed in relation to her dietary adherence with serological and initial symptomatic improvement. However she continued to have periodic episodes of abdominal pain and distension and diarrhoea, affecting both her work and the quality of her home life.
 
The dietary analysis of her food intake, which included a written and photographic food diary, indicated Mrs X was rigorously adhering to a gluten-free diet. After discussion with her consultant, a supersensitive GFD was initiated (avoidance of codex wheat-starch, gluten-free oats, barley malt and all of their derivatives) however this additional restriction also failed to produce significant improvements with her gastrointestinal symptoms.
 
A further consultation with her gastroenterology consultant was arranged, and they suggested a repeat gastroscopy. This indicated an almost complete recovery of Mrs X’s duodenal mucosa with only some raised intraepithelial lymphocytes present demonstrating an improvement from a Marsh grade 3c to 1. Mrs X’s serological and intestinal recovery suggested her on-going symptoms may be functional in nature.
 
It is now well established in the literature that coeliac disease, irritable bowel syndrome (IBS) and other functional bowel disorders are not mutually exclusive and may co-present in the same patient.1 A recent prospective cohort study in patients with coeliac disease suggested the prevalence of some type of functional symptoms at one year follow-up was 47% - despite adherence to a GFD.2 Furthermore, in one RCT, the low FODMAP diet (LFD) has been demonstrated to be superior than further gluten restriction for CD patients in serological and histological remission, with on-going functional symptoms.3
 
Mrs X was reassured by the results of the repeat biopsy, and keen to investigate the LFD as a treatment option. At a dietetic follow-up appointment a baseline of her current symptoms was established with the Gastrointestinal Symptom Rating Scale (GSRS - a validated IBS symptom questionnaire). 4 The principles of the LFD were discussed in detail, Mrs X was provided with sample menus and suitable digital applications were also suggested to facilitate her adoption of the approach. Contact information for the dietitian was provided and Mrs X was encouraged to update us on her progress.
 
Mrs X completed six weeks of the LFD with significant improvement in abdominal pain, distension and stool consistency (see table 2).  A reintroduction phase was discussed and initiated which highlighted particularly lactose, fructose and polyols as dietary triggers for her symptoms. Personalising a liberalised LFD required taking into account her need for a strict gluten-free diet. These two dietary therapies should not be considered synonymous, particularly it was important to insure that FODMAP restrictions did not negatively impact her intake of fibre, calcium and other nutrients.
 
Interestingly Mrs X did report finding the LFD "very socially restrictive" and “difficult to fit with family meals” - this was in addition to her gluten restriction. Some studies have suggested a possible negative effect on health-related quality of life (HRQOL) in implementing the elimination phase of the LFD. Combining this approach with a strict gluten free diet should only be used in patients with proven mucosal recovery, with who IBS and functional symptoms are particularly problematic.


 

Table 2

Key:  Pre LFD. Post LFD
Symptom None Mild Moderate Severe
Abdominal pain    
Abdominal bloating    
Increased flatulence    
Belching or burping      
Abdominal gurgling    
Urgency to open bowels    
Incomplete evacuation      
Nausea      
Heartburn      
Acid regurgitation      
Tiredness/lethargy    
 
Bowels Open 2-5 x / day 2 x / day
Bristol Stool Chart 6-7 5-3
 

References

  1. Sainsbury A, Sanders DS, Ford AC. Prevalence of irritable bowel syndrome-type symptoms in patients with celiac disease: a meta-analysis. Clin Gastroenterol Hepatol. 2013;11(4):359-65.e1. doi:10.1016/j.cgh.2012.11.033.

  2. Silvester JA, Graff LA, Rigaux L, et al. Symptoms of Functional Intestinal Disorders Are Common in Patients with Celiac Disease Following Transition to a Gluten-Free Diet. Dig Dis Sci. July 2017:1-6. doi:10.1007/s10620-017-4666-z.

  3. Nuland K. The effect of a low FODMAP diet in addition to a gluten free diet on symptoms and quality of life in patients with coeliac disease and IBS-like symptoms: A randomized, controlled clinical study NUCLI395 – Master’s Thesis in Clinical Nutrition. http://bora.uib.no/bitstream/handle/1956/13025/144679742.pdf?sequence=1&isAllowed=y. Accessed July 16, 2017.

  4. Svedlund J, Sjödin I, Dotevall G. GSRS--a clinical rating scale for gastrointestinal symptoms in patients with irritable bowel syndrome and peptic ulcer disease. Dig Dis Sci. 1988;33(2):129-134. doi:10.1007/BF01535722.
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