Abstract
BackgroundCeliac disease can present in children and adults with a variety of manifestations including a rare complication known as ulcerative jejunitis. The latter has been associated with refractory celiac disease in adult onset patients. The objective of this case report is to describe the first pediatric case of ulcerative jejunitis in celiac disease, diagnosed by capsule endoscopy, which was not associated with refractory celiac disease.Case presentationThe 9 year old girl presented with a history of abdominal pain and vomiting. Laboratory investigations revealed a slightly elevated IgA tissue transglutaminase antibody level in the setting of serum IgA deficiency. Initial upper endoscopy with biopsies was not conclusive for celiac disease. Further investigations included positive IgA anti-endomysium antibody, and positive HLA DQ2 typing. Video capsule endoscopy showed delayed appearance of villi until the proximal to mid jejunum and jejunal mucosal ulcerations. Push enteroscopy with biopsies subsequently confirmed the diagnosis of celiac disease and ulcerative jejunitis. Immunohistochemical studies of the intraepithelial lymphocytes and PCR amplification revealed surface expression of CD3 and CD8 and oligoclonal T cell populations. A repeat capsule study and upper endoscopy, 1 year and 4 years following a strict gluten free diet showed endoscopic and histological normalization of the small bowel.ConclusionUlcerative jejunitis in association with celiac disease has never previously been described in children. Capsule endoscopy was essential to both the diagnosis of celiac disease and its associated ulcerative jejunitis. The repeat capsule endoscopy findings, one year following institution of a gluten free diet, also suggest that ulcerative jejunitis is not always associated with refractory celiac disease and does not necessarily dictate a poor outcome.
Highlights
Celiac disease can present in children and adults with a variety of manifestations including a rare complication known as ulcerative jejunitis
Ulcerative jejunitis in association with celiac disease has never previously been described in children
Capsule endoscopy was essential to both the diagnosis of celiac disease and its associated ulcerative jejunitis
Summary
Ulcerative jejunitis in association with CD has never, based on our review of the literature, been described in the pediatric age group. CE was essential to establishing the initial diagnosis of CD in this patient and in identifying the associated ulcerative jejunitis, as well as its resolution. The CE findings suggest that ulcerative jejunitis is not always associated with RCD, and as in this case, does not necessarily dictate a poor outcome. Further studies are needed to evaluate the incidence and outcome of ulcerative jejunitis and RCD in children with celiac disease. ES: Helped in preparation of manuscript, endoscopist, read the video capsule studies and current primary gastroenterologist. Author details 1Division of Pediatric Gastroenterology, Montreal Children’s Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada. Author details 1Division of Pediatric Gastroenterology, Montreal Children’s Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada. 2Department of Pathology, Montreal Children’s Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada
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