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Open Access Case report

Ulcerative jejunitis in a child with celiac disease

Terry Sigman1*, Van-Hung Nguyen2, Florin Costea1, Ana Sant’Anna1 and Ernest G Seidman1

Author Affiliations

1 Division of Pediatric Gastroenterology, Montreal Children’s Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada

2 Department of Pathology, Montreal Children’s Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada

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BMC Gastroenterology 2014, 14:29  doi:10.1186/1471-230X-14-29

Published: 13 February 2014

Abstract

Background

Celiac 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 presentation

The 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.

Conclusion

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. 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.

Keywords:
Ulcerative jejunitis; Celiac disease; Pediatric; Capsule endoscopy