Celiac disease, collagenous sprue and microscopic colitis in IBD. Observations from a population-based cohort of IBD (ICURE).

OBJECTIVE: Inflammatory bowel disease (IBD), microscopic colitis and celiac disease are all diseases with worldwide distribution and increased incidence has been reported from many areas. There is a shortage of studies investigating the occurrence of these diseases in the same individual and whether those affected demonstrate any particular phenotype. The aim of the study was to describe the concomitant incidence of microscopic colitis and celiac disease in a population-based IBD cohort. METHODS: All 790 individuals in a prospective population-based cohort included 2005-09 from Uppsala region, Sweden, were reviewed regarding the appearance of microscopic or celiac disease before or after IBD diagnosis. RESULTS: Fifty percent (396/790) of the patients had been examined for the possibility of celiac disease. Seventeen patients with celiac disease were found, representing 2.2% of the cohort. Patients with celiac disease were younger compared to the non-celiac patients and those with colitis had more often an extensive inflammation of the colon. Seventy-one percent (12/17) were women. The majority of the patients were diagnosed with celiac disease before IBD. Five patients with IBD had an earlier diagnosis of microscopic colitis or developed it after the IBD diagnosis. One teenager developed collagenous sprue, misinterpreted as a severe relapse of ulcerative colitis (UC) resulting in colectomy. CONCLUSIONS: The risk for celiac disease seems not to be increased in IBD, but those affected by both diseases seem to be predominantly women with extensive UC. There is a potential association between microscopic colitis and IBD.

Collagenous sprue in a 3-month-old infant.

Collagenous sprue (CS) is a severe malabsorption disorder, the etiology of which has not been well defined. Herein, we report the case of a 3-month-old infant with CS who responded to steroid and immunomodulator treatment and presented a thick subepithelial collagen band. A 3-month-old Japanese girl presented with severe watery diarrhea that lasted for 2 weeks. She was admitted to the referring hospital, but symptomatic improvement was not achieved with fasting and rehydration. Gastroduodenal endoscopy showed an edematous duodenal mucosal surface. Duodenal biopsy indicated severe villous atrophy with infiltration of mostly CD8-positive T cells; and deposition of subepithelial collagen was confirmed. The subepithelial collagen deposits, however, had disappeared after treatment. Historically, child-onset CS is extremely rare and this case is likely to be the youngest case of infantile CS. The present case suggests that CS should be considered as a differential diagnosis for intractable diarrhea, even in infants.

A rare cause of protein losing enteropathy: collagenous sprue.

Collagenous sprue is a clinicopathological entity with an unknown etiology. Its clinical features include progressive malabsorption, diarrhea, weight loss, unresponsiveness to treatment, and high mortality rates. The age interval of collagenous sprue is quite broad and ranges between 2 and 85 years. As far as to our knowledge, the presented case is the first reported case in infancy.

Collagenous sprue diagnosed by double balloon endoscopy.

Double balloon endoscopy (DBE) is useful for diagnosing many intestinal diseases and for endoscopic procedures. We report a case of chronic diarrhea in a 58-year-old Japanese man. He was initially suspected to have malabsorption syndrome. DBE showed reduction of folds, scalloping, mucosal nodularity and granularity. Pathological examinations of biopsies from the jejunum showed severe villous atrophy with subepithelial collagen bands. These findings led to the final diagnosis of collagenous sprue (CS). With1 month of total parenteral nutrition followed by a low-gluten diet, his symptoms gradually improved. CS has never been reported before in Japan. DBE is useful for making a diagnosis of CS, and may be considered for patients who are suffering from diarrhea of unknown cause.

[Collagenous sprue: secondary or independent from celiac disease?]. / Esprúe colágeno: ¿entidad secundaria o independiente de la enfermedad celíaca?

Collagenous sprue is a rare disease that goes with persistent diarrhea, weight loss and bad absortion, because it affects the small intestine, mainly duodenum and proximal jejunum. Diagnosis is made by having clinical signs and histological proof of atrophy and subepitelial deposit of collagenous material. Its etiology is not known completely, it is proposed that the origin is autoimmune because its relationship with celiac disease. Also there is a proposal that is a celiac evolution to gluten free diet. Is because this is not clear that we present a case of a patient with bad absorptive diarrhea and a clinical expression of collagenous sprue, that had a great clinical response to corticosteroids with home parenteral nutrition center.

Collagenous sprue: don´t forget connective tissue in chronic diarrhea evaluation.

Collagenous sprue is a rare disease of the small bowel characterized by mucosal atrophy and excessive subepithelial collagen deposition. The etiology remains unclear and the diagnosis is based upon patient´s clinical picture and anatomopathological findings. Clinically, collagenous sprue is characterized by persistent diarrhoea, severe malabsorption, multiple nutrient deficiencies and progressive weight loss. Differential diagnosis includes celiac disease, which is mandatory to rule out because of their frequent association. Glutenfree diet is the first therapeutic step, but it usually is not effective. However, recent studies show high success rates with immunomodulators, mainly corticosteroids. We report the case of a patient presenting with chronic diarrhea and severe malabsorption who was diagnosed with collagenous sprue, with no response to gluten free diet, but with excellent response to budesonida.

An elderly lady with significant weight loss.

We present a rare case of collagenous sprue in an elderly woman with significant weight loss and malnutrition. Collagenous sprue is a rare, female-predominant and immune-mediated gastrointestinal disease that can affect any part of the gut, and shares a strong association with Coeliac disease. The diagnosis is confirmed by gut histopathology demonstrating a subepithelial collagenous band and inflammatory infiltrate in the lamina propria. The pathogenesis and natural history is poorly elucidated, and treatment involves a gluten-free diet and/or immunomodulatory therapy.

Collagenous enterocolitis manifesting as watery diarrhoea and iron-deficiency anaemia.

A 56-year-old Caucasian woman presented with epigastric pain, watery diarrhoea, bloating and flatulence following treatment with duloxetine and venlafaxine for anxiety and depression. Abdominal examination was benign. Blood work revealed haemoglobin of 96 g/L (115-160 g/L), iron 6 µmol/L (10-33 µmol/L), transferrin saturation 0.08 (0.20-0.55), ferritin 26 µg/L (15-180 µg/L), albumin 46 g/L (35-50 g/L), pre-albumin 293 mg/L (170-370 mg/L), total IgA 2.64 g/L (0.78-3.58 g/L) and anti-tTG IgA 5 units (<20 units). Faecal occult blood tests were 3/3 positive and stool cultures were negative. CT enterography was normal. Colonic biopsy revealed collagenous colitis, while duodenal biopsy showed collagenous sprue with blunted to completely flattened villi and markedly thickened subepithelial collagen table entrapping capillaries and lymphocytes. The patient started a gluten-free diet, loperamide and ferrous gluconate. Her symptoms resolved and a faecal immunochemical test performed 6 months later was negative.

Collagenous sprue: a coeliac disease look-alike with different treatment strategy.

Collagenous sprue is a rare clinicopathological condition of the small bowel. It is characterised by abnormal subepithelial collagen deposition and is typically associated with malabsorption, diarrhoea and weight loss. The clinical features of collagenous sprue often resemble those of coeliac disease and together with frequent histological findings like mucosal thinning and intraepithelial lymphocytosis the diagnosis may be hard to reach without awareness of this condition. While coeliac disease is treated using gluten restriction, collagenous sprue is, however, not improved by this intervention. In cases of diet-refractory 'coeliac disease' it is therefore essential to consider collagenous sprue to initiate treatment at an early stage to prevent the fibrotic progression. Here, we report a case of a 78-year-old man with collagenous sprue and present the clinical and histological manifestations as well as the successful treatment course that he underwent.

Proximal collagenous gastroenteritides: clinical management. A systematic review.

AIM: While collagenous colitis represents the most common form of the collagenous gastroenteritides, the collagenous entities affecting the proximal part of the gastrointestinal tract are much less recognized and possibly overlooked. The aim was to summarize the latest information through a systematic review of collagenous gastritis, collagenous sprue, and a combination thereof. METHOD: The search yielded 117 studies which were suitable for inclusion in the systematic review. Excluding repeated cases, 89 case reports and 28 case series were reported, whereas no prospective studies with or without control groups were identified. Further, no randomized, controlled trials were identified. The total number of patients with proximal collagenous gastroenteritides reported was 330. RESULTS: An overview of clinical presentations, prognosis, pathophysiology and histopathology, as well as management of these disorders is presented. The prognosis of both collagenous gastritis and sprue seems not to be as dismal as considered previously. Data point to involvement of immune or autoimmune mechanisms potentially driven by luminal antigens initiating the fibroinflammatory condition. CONCLUSIONS: To reach the diagnosis it is recommended that biopsies are obtained during gastroduodenoscopies. Therapies with anti-secretory strategies, glucocorticoids, and in some cases iron supplementation are suggested, although rational treatment options from randomized, controlled trials do not exist for these rare or even overlooked disorders.

Angiotensin-II inhibitor (olmesartan)-induced collagenous sprue with resolution following discontinuation of drug.

Collagenous sprue (CS) is a pattern of small-bowel injury characterized histologically by marked villous blunting, intraepithelial lymphocytes, and thickened sub-epithelial collagen table. Clinically, patients present with diarrhea, abdominal pain, malabsorption, and weight loss. Gluten intolerance is the most common cause of villous blunting in the duodenum; however, in a recent case series by the Mayo Clinic, it has been reported that olmesartan can have a similar effect. In this case report, a 62-year-old female with a history of hypothyroidism and hypertension managed for several years with olmesartan presented with abdominal pain, weight loss, and nausea. Despite compliance to a gluten-free diet, the patient's symptoms worsened, losing 20 pounds in 3 wk. Endoscopy showed thickening, scalloping, and mosaiform changes of the duodenal mucosa. The biopsy showed CS characterized by complete villous atrophy, lymphocytosis, and thickened sub-epithelial collagen table. After 2 mo cessation of olmesartan, the patient's symptoms improved, and follow-up endoscopy was normal with complete villous regeneration. These findings suggest that olmesartan was a contributing factor in the etiology of this patient's CS. Clinicians should be aware of the possibility of drug-induced CS and potential reversibility after discontinuation of medication.

Atrofia vellositaria sin enfermedad celíaca: ¿un nuevo síndrome o más confusión? / Non celiac villous atrophy: More confusion or a new syndrome?

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