A general health economics review of the hidden costs involved in discharging coeliac patients from hospital-based specialty clinics to community-based management.

Aim: The aim of this work was to highlight the impact and hidden costs incurred by the NHS in supporting this management process. Background: Coeliac disease (CD) is a common auto-immune condition which affects around 1% of the general population. In 2005 there was a drive by the government to discharge patients with CD from specialist hospital follow up to community-based management to improve cost efficiency. Methods: A retrospective analysis of 1317 CD patients collected from a local coeliac database created between 2005 and 2016. Results: During these 12 years, CD patients accounted for 1965 hospital admissions with a total 5716 days spent within the hospital setting. There were 33150 adult and paediatric OPAs attended equating to 25.17 per coeliac patient, or 2.29 per person per year. The cost to the CCG totalled £5,167,396. A total of 527 lower GI procedures were undertaken with findings of microscopic colitis, melanosis coli, inflammatory bowel disease and colon cancer. 420 (29%) of the coeliac cohort were found to have IDA with just 4% (17/420) receiving an intravenous (IV) iron infusion. Conclusion: It would appear that the government's attempts to reduce the cost of CD care within the NHS was not particularly effective, from a financial, or patient care perspective. A hospital-based, specialist nurse led, virtual management system (with consultant over-view) may prove to be a more efficient compromise, to help reduce down waiting times and costs, whilst still providing coeliac patients with the specialist and holistic input they require and deserve.

Quality of Diet of Patients with Coeliac Disease in Comparison to Healthy Children.

A lifelong strict gluten-free diet is the only available treatment for patients with coeliac disease (CD). As with any restrictive diet, it may potentially lead to nutritional deficits. Seventy-six patients with CD (mean age 9.0 ± 4.3 years, 57% female) and 590 healthy controls (HC) (mean age 9.9 ± 0.1 years, 54% female) were recruited and requested to keep a 3-day food record (3DFR). In HC patients, anthropometric data were determined at the time when 3DFRs were collected. In CD patients, anthropometric data were determined at two time points: at diagnosis and at the time of 3DFRs collection. Intake of energy, macronutrients, and micronutrients was determined using PRODI expert 6.9 software and expressed as a percentage of recommended daily intake. In CD patients, all measured anthropometric measures (body weight (BW), body height (BH), and body mass index (BMI) z-scores) increased significantly after the mean duration of 34.1 months of a GFD. Overall, CD patients had significantly lower BW and BH z-scores compared to healthy controls. Patients with CD were generally more compliant with the recommended dietary intakes of macronutrients and some micronutrients, as compared to HC. Three participants were not compliant with the GFD; 42.1% of participants took oral nutritional supplements at the start of their GFD. Our study showed that patients with CD have better compliance with dietary recommendations compared to healthy controls, showing that a well-balanced GFD diet can provide necessary macro- and micronutrients.

Development of age-dependent micronutrient centile charts and their utility in children with chronic gastrointestinal conditions at risk of deficiencies: A proof-of-concept study.

BACKGROUND & AIMS: Interpretation of blood micronutrient levels requires age-appropriate reference intervals. This study developed age-dependent micronutrient centiles for healthy children (HC) and explored their utility in sick children. METHODS: 244 blood samples were collected from normal HC who underwent tests for acute illness. Age-dependent, centile charts were fitted for zinc, copper, magnesium and selenium in plasma and erythrocytes (RBC), and for vitamins B1, B2 and B6 in RBC. For 34 children with Crohn's disease (CrD) and 55 with coeliac disease (CoeD), Z-scores for the levels of these micronutrients were computed, using the new charts. Associations were explored between plasma and RBC micronutrient Z-scores, and in CrD with CRP and serum albumin. RESULTS: In HC, plasma zinc and selenium increased and plasma copper, magnesium and RBC vitamins B1, B2 and B6 decreased with age. In HC and in CrD, plasma and RBC Z-scores for copper, selenium and magnesium (all p < 0.001) were positively correlated, but not for zinc. In CrD, albumin was related with plasma zinc (rho = 0.62; p < 0.001) and selenium Z-scores (rho = 0.65; p < 0.001) and plasma copper Z-score with CRP (rho = 0.45; p = 0.02). A higher proportion of CrD children had low levels for B2 (21% vs 0%; p = 0.01) and B6 (18% vs 0%; p = 0.02) using the new centile charts than the local laboratory references. CONCLUSION: Age-dependent micronutrient centile charts enable tracking of micronutrient status, Z-score calculation and may prevent misdiagnosis and inappropriate treatment of deficiencies. In systemic inflammatory conditions, RBC measurements of certain micronutrients may be more reliable to use than measurements in plasma.

Gluten-free diet adherence and implications for the diagnosis of coeliac disease.

Coeliac disease (CD) is an autoimmune disorder caused by the ingestion of gluten containing foods in genetically susceptible individuals, with a worldwide prevalence of up to 1%. Currently, the only available treatment is a gluten-free diet (GFD). Screening for CD is primarily performed using serum based testing for anti-tissue transglutaminase (tTG) antibodies. Patients must be on a gluten containing diet at the time of testing to ensure an accurate serological result. We investigated the prevalence of a GFD in hospital clinic settings and the general population using survey data to estimate the proportion of CD patients that may be misdiagnosed for CD based on serological tests. Data were collected at clinics of a metropolitan hospital in Sydney, Australia, and the general population. Data from Medicare Benefits Scheme and tTG results from a large Australian private laboratory were reviewed for comparison. Of 778 participants who responded to the survey, 58 (7.5%) were on a GFD. More patients attending the immunology (15.9%) and gastroenterology (12.1%) clinics adopted a GFD than those attending the diabetes (2.6%) or endocrinology (6.1%) clinics, or in the general population (4.3%). More females than males excluded gluten from their diet (p<0.0001). Medicare statistics between 2013 and 2019 demonstrated an increase in CD serological testing; however, tTG data from a private pathology highlighted a stable level of elevated tTG antibodies of 3% of total tests performed. The high number of individuals on a GFD is likely impacting the ability to accurately diagnose CD using serum-based testing.

Prevalence and management of vitamin D deficiency in children with newly diagnosed coeliac disease: cohort study.

BACKGROUND: Coeliac disease (CD) causes deficiency of various micronutrients including vitamin D, and there are no specific guidelines for treatment. AIMS: To determine the prevalence of vitamin D deficiency in children newly diagnosed with CD and the role of oral high-dose vitamin D in its treatment. METHODS: Calcium intake, sun exposure and biochemical and radiological parameters related to vitamin D deficiency were compared between 60 children aged 0-18 years diagnosed with CD and 60 healthy age- and sex-matched controls. The cases with serum 25(OH)D (<20 ng/ml) were given oral vitamin D (60,000 IU/week) and calcium (500 mg/day) for 12 weeks, along with a gluten-free diet (GFD); they were re-evaluated within a week of completion. The primary outcome measure was the serum 25(OH)D level, and secondary measures included serum calcium, phosphorus, alkaline phosphatase, parathormone and clinical and/or radiological rickets. RESULTS: The prevalence of vitamin D deficiency (25(OH)D <20 ng/ml) was significantly greater in the cases (n=38, 63.3%) than in the controls (n=27, 45.0%). Upon treatment, all 38 cases with vitamin D deficiency showed a significant rise in 25(OH)D levels along with normalisation of other biochemical abnormalities. Two children had 25(OH)D levels >100 ng/ml with no other feature suggestive of vitamin D toxicity. CONCLUSIONS: Vitamin D deficiency is more prevalent in children with CD. Administration of oral high-dose vitamin D for 12 weeks along with a GFD leads to a robust response, indicating rapid mucosal recovery. The vitamin D dosage recommended for malabsorption states may be excessive in CD.Abbreviations: ALP: alkaline phosphatase; CaBP: calcium-binding proteins; CD: coeliac disease; GFD: gluten-free diet; PTH: parathormone; RU: reproducibility units; 25(OH)D: 25 hydroxy vitamin D.

Efficacy and safety of hyperbaric oxygen therapy for moderate-to-severe ulcerative colitis: a protocol for a systematic review and meta-analysis.

INTRODUCTION: Ulcerative colitis (UC) is a type of inflammatory bowel disease, and 62% of patients with UC felt that it is difficult for them to live a normal life. Furthermore, some researches have shown that about 15% of patients with UC undergo at least one extreme clinical course in their lifetime, and 10%-30% of patients with UC oblige colectomy. Although many investigations have demonstrated that HBO2 has a beneficial impact on UC treatment, a systematic review and meta-analysis are unavailable. Therefore, a meta-analysis is essential to assess the efficacy and safety of HBO2 in treating UC. METHODS AND ANALYSIS: A systematic search plan will be performed in the following seven databases with a restriction of time from inception to September 2020 to filter the eligible studies: PubMed, Web of Science, Embase, Cochrane Library, China National Knowledge Infrastructure, Chinese Scientific Journal Database (VIP) and Chinese Biomedical Database WanFang. Other related resources will be also searched. Two independent reviewers will choose eligible researches and extract data. The risk of bias will be evaluated based on Cochrane Collaboration's Risk of Bias tool and Newcastle-Ottawa Scale. Eventually, a systematic review and meta-analysis will be performed via the Review Manager V.5.3 statistical software and STATA V.14.0 software. ETHICS AND DISSEMINATION: This study will not involve the individual patient and any ethical problems since its outcomes are based on published data. Therefore, no ethical review and approval are required. We plan to publish the study in a peer-reviewed journal. PROSPERO REGISTRATION NUMBER: CRD42020210244.

Exposure to the Danish Mandatory Vitamin D Fortification Policy in Prenatal Life and the Risk of Developing Coeliac Disease-The Importance of Season: A Semi Ecological Study.

Few studies have examined the role of maternal diet in relation to development of coeliac disease (CD). In Denmark, cancellation of mandatory vitamin D fortification of margarine in June 1985 provided this opportunity. This study examined if season of birth or prenatal exposure to extra vitamin D from food fortification were associated with developing CD later in life. A strength of this study is the distinctly longer follow-up of patients (30 years). This register-based study has a semi-ecological design. Logistic regression analysis was used to estimate odds ratios and to calculate 95% confidence intervals. The odds ratio for developing CD was 0.81 (95% CI 0.66; 1.00 p = 0.054), comparing those with fetal exposure to mandatory vitamin D fortification policy of margarine to those without after adjusting for gender and season of birth. There was a statistically significant season effect particularly for children born in autumn (OR 1.6 95% CI 1.16; 2.21) and born in summer (OR 1.5 95% CI 1.1; 2.1) when compared to children born in winter. Although this study did not find evidence to support the premise that prenatal exposure to small extra amounts of vitamin D from a mandatory food fortification policy lowered risk of developing CD, the small number of CD cases and observed association between season of birth and CD suggest that environmental exposure ought to be further explored.

Comparing alternative follow-up strategies for patients with stable coeliac disease.

BACKGROUND: Once clinically stable, patients with coeliac disease should have annual follow-up. Lack of capacity in gastroenterology outpatient clinics mean alternatives are required. OBJECTIVES: We studied the effectiveness of follow-up deferred to general practitioners (GP-FU) and compared this with a neighbouring Trust where follow-up was through a dedicated nurse-led telephone clinic (T-FU). DESIGN: All patients with coeliac disease were posted a questionnaire examining patient satisfaction, adherence with gluten-free diet and calcium intake. RESULTS: 517 of 825 patients (62.7%) completed a postal questionnaire (median age 61, 72% female). 28% of GP-FU and 84% of T-FU patients received an annual review. Of those seen, 33% (GP-FU) and 53% (T-FU) were weighed (χ2 65.8, p<0.001), 44% and 63% had symptom review (χ2 81.1, p<0.001) and 33% and 51% had dietary adherence checked (χ2 60.6, p<0.001). Almost all patients considered their adherence with gluten-free diet (GFD) good or excellent, although the majority of patients failed to achieve the recommended daily intake of calcium. GP-FU patients were more likely to receive calcium±vitamin D supplements (77% vs 42%, χ2 88.2, p<0.001) and they were also more likely to receive appropriate vaccinations (67% vs 38%, χ2 17.6, p<0.001). CONCLUSIONS: Discharge of patients with coeliac disease to primary-care in many cases results in their complete loss to follow-up. When patients were reviewed, either by GP-FU and T-FU, many aspects of their care are not addressed. Whether this will result in late complications remains to be seen.

Persistent Myocardial Ischaemia due to Anaemia in a Patient with Coeliac Disease - A Case Report.

INTRODUCTION: Coeliac disease (CD) is a systemic disease of inappropriate immune response to gluten, and is associated with 10% increased risk of cardiovascular disease. Here we present a case of a young patient with persistent myocardial ischaemia due to iron-deficiency anaemia despite oral iron supplementation, who was eventually diagnosed with CD. CASE REPORT: A 36-year-old man was admitted to the cardiology department due to ST-elevation myocardial infarction of the inferior wall. Emergency coronary angiography showed occlusion of the right coronary artery and intermediate-diameter lesions in other arteries. Primary percutaneous coronary intervention with stent implantation to the right coronary artery was performed. Despite the successful intervention, the patient presented with recurrent chest pain, persistent tachycardia up to 120 beats per minute and syncope. Coronary angiography was repeated and the intermediate-diameter lesions were proved insignificant. Laboratory tests revealed microcytic anaemia with haemoglobin level of 6.5 g/dL, despite oral iron supplementation in the pre-hospital period. An emergency gastroscopy with duodenoscopy revealed flat duodenal mucosa. The duodenal biopsy confirmed the diagnosis of CD. Strict gluten-free diet and further iron supplementation were recommended. Two months later the patient presented with no recurrent chest pain and normal haemoglobin concentration. CONCLUSIONS: Diagnosis of CD with atypical presentation can be difficult. Iron-deficiency anaemia refractory to oral iron supplementation should always raise the suspicion of CD, even without typical gastrointestinal manifestation.

The evolution of celiac disease publications: a holistic approach with bibliometric analysis.

BACKGROUND: Despite the increasing number of publications on celiac disease, there is a lack of studies that made a holistic bibliometric evaluation of the studies on this topic. AIMS: The purpose of this study is to analyze the publications about celiac disease by using bibliometric methods and this way to demonstrate the celiac disease-related trends, top effective articles, journals, and international collaborations between the countries and institutions. METHODS: All articles published between 1980 and 2018 on celiac disease were downloaded from Web of Science and analyzed with bibliometric methods. The correlations between economic development and publication productivity of the countries were investigated with Spearman's rank correlation coefficient. Linear regression analysis was used to estimate the number of publications and citations. RESULTS: The literature review showed that there were 6545 articles about celiac disease published between the years 1980 and 2018. The top productive country that produced most publications about celiac disease was the Italy. The most prolific journals were the Journal of Pediatric Gastroenterology and Nutrition and Scandinavian Journal of Gastroenterology. There was a moderate positive significant correlation between the number of publications and gross domestic product (r = 0.639, p < 0.001). CONCLUSIONS: Despite the advanced research on the diagnosis, pathophysiology, and treatment of celiac disease, the global research level about the disease is low. Therefore, the international collaboration about the disease should not remain at only regional contexts; the exchange of knowledge and common studies especially in developing or underdeveloped countries should be supported in terms of prevalence and clinic studies.

European Society for the Study of Coeliac Disease (ESsCD) guideline for coeliac disease and other gluten-related disorders.

This guideline presents recommendations for the management of coeliac disease (CD) and other gluten-related disorders both in adults and children. There has been a substantial increase in the prevalence of CD over the last 50 years and many patients remain undiagnosed. Diagnostic testing, including serology and biopsy, should be performed on a gluten-containing diet. The diagnosis of CD is based on a combination of clinical, serological and histopathological data. In a group of children the diagnosis may be made without biopsy if strict criteria are available. The treatment for CD is primarily a gluten-free diet (GFD), which requires significant patient education, motivation and follow-up. Slow-responsiveness occurs frequently, particularly in those diagnosed in adulthood. Persistent or recurring symptoms necessitate a review of the original diagnosis, exclude alternative diagnoses, confirm dietary adherence (dietary review and serology) and follow-up biopsy. In addition, evaluation to exclude complications of CD, such as refractory CD or lymphoma, should be performed. The guideline also deals with other gluten-related disorders, such as dermatitis herpetiformis, which is a cutaneous manifestation of CD characterized by granular IgA deposits in the dermal papillae. The skin lesions clear with gluten withdrawal. Also, less well-defined conditions such as non-coeliac gluten sensitivity (NCGS) and gluten-sensitive neurological manifestations, such as ataxia, have been addressed. Newer therapeutic modalities for CD are being studied in clinical trials but are not yet approved for use in practice.

Analysis of ingredient and nutritional labeling of commercially available gluten-free bread in Brazil.

This study aimed at evaluating the ingredients and nutritional information of commercially-available gluten-free bread (GFB) in Brazil. A total of 128 products were studied, of which 87% presented the sandwich loaf shape. Traditional GFBs (n = 114) had as main ingredient the refined rice flour and starches, whereas alternative ones (n = 14) presented whole rice flour. Raw materials suggested by science to improve nutrients and bioactive compounds of gluten-free foodstuffs were observed in the ingredient list of most products (n = 86); however, they were used in lower levels, thus no significant differences were observed for nutritional information between the different categories of GFB. No products with added vitamins or minerals were found, though 77% of them included hydrocolloids in their formulations - other food additives were also observed. Despite the increased gluten-free food market, there is still a gap between science and market, especially regarding the approaches to improve the GFB diversity and nutritional quality.

Prevalence of reduced bone mineral density in adults with coeliac disease - are we missing opportunities for detection in patients below 50 years of age?

OBJECTIVE: There are little data on the prevalence of reduced bone mineral density (BMD) in young adult patients with coeliac disease; guidelines do not support routine investigation of these patients. We assessed the prevalence of reduced BMD in our patients by age. PATIENTS AND METHODS: Prospective observational study of 260 coeliac patients having DXA one year after commencing gluten-free diet. Nonparametric tests and regression were used. RESULTS: Median age was 51years, BMI 24 and 85 (32.7%) were male. Reduced BMD was associated with increasing age (p < .001), female sex (p = .005), low BMI (p < .001) and previous fracture (p < .01); 49% of all patients and all patients under 20 years old had reduced BMD. The median age of patients with BMI <20 kgm2 was 56 (27, 70) years with the majority of younger patients having normal BMI. CONCLUSIONS: Low BMD is a common finding in young patients with coeliac disease, yet routine assessment of BMD is not currently supported by national guidelines. Early identification may improve motivation to comply with GFD and allow adequate calcium and vitamin D supplementation to reduce risk of fracture later in life.

The Availability and Nutritional Adequacy of Gluten-Free Bread and Pasta.

Management of coeliac disease (CD) requires the removal of gluten from the diet. Evidence of the availability, cost, and nutritional adequacy of gluten-free (GF) bread and pasta products is limited. GF flours are exempt from UK legislation that requires micronutrient fortification of white wheat flour. This study surveyed the number and cost of bread and pasta products available and evaluated the back-of-pack nutritional information, the ingredient content, and the presence of fortification nutrients of GF bread and pasta, compared to standard gluten-containing equivalent products. Product information was collected from four supermarket websites. Standard products were significantly cheaper, with more products available than GF (p < 0.05). GF bread products were significantly higher in fat and fiber (p < 0.05). All GF products were lower in protein than standard products (p < 0.01). Only 5% of GF breads were fortified with all four mandatory fortification nutrients (calcium, iron, niacin, and thiamin), 28% of GF breads were fortified with calcium and iron only. This lack of fortification may increase the risk of micronutrient deficiency in coeliac sufferers. It is recommended that fortification legislation is extended to include all GF products, in addition to increased regulation of the nutritional content of GF foods.

Risk of Fractures in Youths with Celiac Disease-A Population-Based Study.

OBJECTIVE: To assess the risk of any fracture requiring hospital care in a cohort of individuals with celiac disease diagnosed in childhood/adolescence compared with reference individuals matched by age and sex. STUDY DESIGN: Our study cohort consisted of 213 635 people born and residing in Friuli-Venezia Giulia Region, Italy, in 1989-2011. We selected, through pathology reports, hospital discharge records, or co-payment exemptions, 1233 individuals with celiac disease (aged 0-17 years at diagnosis) and compared them with 6167 reference individuals matched by sex and year of birth. Fractures were identified through hospital discharge records. We calculated hazard ratios (HRs) for any fracture after celiac disease diagnosis (or index date for reference individuals) with Cox regression and ORs for any fracture before celiac disease diagnosis with conditional logistic regression. RESULTS: During the follow-up period (maximum 23 years), 22 individuals with celiac disease (9394 person-years) and 128 reference individuals (47 308 person-years) experienced a fracture, giving an overall HR of 0.87 (95% CI 0.55-1.37). The risk was not modified by sex, age at diagnosis, or calendar period of diagnosis. We obtained similar HRs when excluding fractures occurring after the age of 18 years and adjusting for maternal education or vitamin D supplementation. The odds of previous fracture also did not differ between subjects with celiac disease and reference individuals (22 and 96 cases, respectively: OR 1.15; 95% CI 0.72-1.84). CONCLUSIONS: We did not find any evidence of an increased risk of fractures during childhood and youth among patients with celiac disease.

Trends, geographical variation and factors associated with prescribing of gluten-free foods in English primary care: a cross-sectional study.

OBJECTIVES: There is substantial disagreement about whether gluten-free foods should be prescribed on the National Health Service. We aim to describe time trends, variation and factors associated with prescribing gluten-free foods in England. SETTING: English primary care. PARTICIPANTS: English general practices. PRIMARY AND SECONDARY OUTCOME MEASURES: We described long-term national trends in gluten-free prescribing, and practice and Clinical Commissioning Group (CCG) level monthly variation in the rate of gluten-free prescribing (per 1000 patients) over time. We used a mixed-effect Poisson regression model to determine factors associated with gluten-free prescribing rate. RESULTS: There were 1.3 million gluten-free prescriptions between July 2016 and June 2017, down from 1.8 million in 2012/2013, with a corresponding cost reduction from £25.4 million to £18.7 million. There was substantial variation in prescribing rates among practices (range 0 to 148 prescriptions per 1000 patients, IQR 7.3-31.8), driven in part by substantial variation at the CCG level, likely due to differences in prescribing policy. Practices in the most deprived quintile of deprivation score had a lower prescribing rate than those in the highest quintile (incidence rate ratio 0.89, 95% CI 0.87 to 0.91). This is potentially a reflection of the lower rate of diagnosed coeliac disease in more deprived populations. CONCLUSION: Gluten-free prescribing is in a state of flux, with substantial clinically unwarranted variation between practices and CCGs.

A randomised, double-blind, cross-over trial to evaluate bread, in which gluten has been pre-digested by prolyl endoprotease treatment, in subjects self-reporting benefits of adopting a gluten-free or low-gluten diet.

The aim of the present study was to determine if the enzyme Aspergillus niger prolyl endoprotease (ANPEP), which degrades the immunogenic proline-rich residues in gluten peptides, can be used in the development of new wheat products, suitable for gluten-sensitive (GS) individuals. We have carried out a double-blind, randomised, cross-over trial with two groups of adults; subjects, self-reporting benefits of adopting a gluten-free or low-gluten diet (GS, n 16) and a control non-GS group (n 12). For the trial, volunteers consumed four wheat breads: normal bread, bread treated with 0·8 or 1 % ANPEP and low-protein bread made from biscuit flour. Compared with controls, GS subjects had a favourable cardiovascular lipid profile - lower LDL (4·0 (sem 0·3) v. 2·8 (sem 0·2) mmol/l; P=0·008) and LDL:HDL ratio (3·2 (sem 0·4) v. 1·8 (sem 0·2); P=0·005) and modified haematological profile. The majority of the GS subjects followed a low-gluten lifestyle, which helps to reduce the gastrointestinal (GI) symptoms severity. The low-gluten lifestyle does not have any effect on the quality of life, fatigue or mental state of this population. Consumption of normal wheat bread increased GI symptoms in GS subjects compared with their habitual diet. ANPEP lowered the immunogenic gluten in the treated bread by approximately 40 %. However, when compared with the control bread for inducing GI symptoms, no treatment effects were apparent. ANPEP can be applied in the production of bread with taste, texture and appearance comparable with standard bread.

Rhabdomyolysis and coeliac disease: A causal or casual association? A case report and review of literature.

BACKGROUND: Rhabdomyolysis is a rare, potentially life-threatening condition, caused by multiple disorders. The association with Coeliac Disease (CD) has been rarely reported and in these cases muscular damage was imputed to hypokalemia. Herein we describe a new case of severe rhabdomyolysis in a child subsequently diagnosed as affected by CD, and review previous reports. CASE PRESENTATION: A 3-year-old boy was referred for diarrhea, brown urine, muscular pain/weakness, and no history of muscular trauma. At entry, laboratory tests showed elevated levels of creatine kinase (CK) (x100 unv) and aspartate aminotransferase (AST) (x10 unv), alanine aminotrasferase (ALT) (x5 unv); electrolytes were within the reference range. Twenty-four hours after admission serum CK peaked 115,000 U/L and transaminases increased up to 30 times unv. Hyperhydration treatment was started with renal function monitoring. Urine output decreased little, while serum creatinine and urea nitrogen stayed within the reference range. Serum potassium levels went down to 2.8 mEq/L at day 3, in spite of supplementation. The patient completely recovered at day 16. Main metabolic causes of rhabdomyolysis were ruled out by appropriate tests. Because of rarely reported cases of CD/rhabdomyolysis, anti-tissue transglutaminase (tTG) antibodies were measured and found positive (IgA 34 U/mL, unv <9). HLA typing was DQA1 05:02, DQB1 03:02. As jejunal biopsy showed patchy villous atrophy, gluten free diet (GFD) was prescribed. One year after starting GFD, histology was normal. REVIEW OF LITERATURE: Literature (search engines: PUB MED and GOOGLE SCHOLAR) from 1980 to 2016 retrieved 8 cases (age range: 12 to 75 years old) previously described. CONCLUSION: The present case suggests to check for CD in children with severe rhabdomyolysis. Because severe rhabdomyolysis itself may elevate the serum potassium levels, hypokalemia might go unrecognized as the cause of muscular damage.

Iron deficiency anaemia in a coeliac: a cause for concern?

A 70-year-old woman with a 15-year history of coeliac disease was admitted for treatment and investigation of symptomatic anaemia. Of note, she was recently commenced on aspirin therapy for retinal artery occlusion. This followed a normal gastro-duodenoscopy, colonoscopy and CT abdomen as workup for iron deficiency anaemia. On this occasion, the patient was further investigated with small bowel capsule endoscopy. This revealed an ulcerated lesion in her proximal jejunum suspicious for malignancy, namely lymphoma. A biopsy of the lesion confirmed adenomatous changes and high-grade dysplasia without malignant changes. Given the endoscopic appearances of the lesion, she was further evaluated with a small bowel MRI. This revealed a 3 cm lesion with associated lymphadenopathy but no distant metastases. She proceeded to resection of her small bowel which confirmed an invasive adenocarcinoma of her proximal jejunum. She is currently undergoing adjuvant chemotherapy on an outpatient basis.

The effect of oligofructose-enriched inulin supplementation on gut microbiota, nutritional status and gastrointestinal symptoms in paediatric coeliac disease patients on a gluten-free diet: study protocol for a pilot randomized controlled trial.

BACKGROUND: A lifelong gluten-free diet (GFD) is regarded as the only proven and accepted therapy for coeliac disease (CD). However, even patients who strictly follow a GFD often suffer from intestinal symptoms and malabsorption. Selective modulation of intestinal microbiota with prebiotics could remedy various symptoms associated with CD. The use of prebiotics in the treatment of intestinal diseases remains insufficiently investigated. To our knowledge, this study makes the first attempt to evaluate the effect of prebiotic supplementation on gastrointestinal symptoms and nutritional status of children with CD. We hypothesized that adherence to a GFD supplemented with oligofructose-enriched inulin (Synergy 1) would deliver health benefits to children suffering from CD without any side effects, and that it would alleviate intestinal inflammation, restore and stabilize gut microbial balance and reverse nutritional deficiencies through enhanced absorption of vitamins and minerals. METHODS: A randomized, placebo-controlled clinical trial was designed to assess the impact of the Synergy 1 on paediatric CD patients following a GFD. We randomized 34 children diagnosed with CD into an intervention group receiving 10 g of the Synergy 1 supplement daily and a placebo group (receiving maltodextrin) during a 12-week nutritional intervention. Selected biochemical parameters, nutritional status and the characteristics of faecal bacteria will be determined in samples collected before and after the intervention. Analysis of vitamins and amino acids concentration in biological fluids will allow to assess the dietary intake of crucial nutrients. The compliance to a GFD will be confirmed by a Food Frequency Questionnaire (FFQ-6) and the analysis of serum anti-tissue transglutaminase and faecal gluten immunogenic peptides (GIP). CONCLUSION: The identification of the beneficial effects of the Synergy 1 supplement on children with CD could have important implications for nutritional recommendations for CD patients and for alleviating the harmful effects of the disease. TRIAL REGISTRATION: ClinicalTrials.gov Registration Number: NCT03064997 .