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Ulcerative jejunitis in a child with celiac disease
Terry Sigman1, Van-Hung Nguyen, Florin Costea
1Division of Pediatric Gastroenterology, Montreal Children's Hospital, Faculty of Medicine, McGill University, 2300 Tupper St, Montreal, Quebec H3H 1P3, Canada. terry.sigman@muhc.mcgill.ca.
Insights
This case report details the first pediatric instance of ulcerative jejunitis in celiac disease, diagnosed via capsule endoscopy. The condition resolved with a gluten-free diet, indicating a favorable prognosis in children.
Area of Science:
- Pediatric Gastroenterology
- Gastrointestinal Endoscopy
- Celiac Disease Pathophysiology
Background:
- Celiac disease presents with diverse manifestations, including ulcerative jejunitis, a rare complication primarily documented in adults with refractory celiac disease.
- This report focuses on the first described pediatric case of ulcerative jejunitis in celiac disease.
Observation:
- A 9-year-old girl with abdominal pain and vomiting, initially inconclusive for celiac disease, underwent capsule endoscopy.
- Video capsule endoscopy revealed delayed villous appearance and jejunal mucosal ulcerations, leading to further diagnostic investigations.
- Immunohistochemical and PCR studies identified specific T cell populations within the jejunal mucosa.
Findings:
- Capsule endoscopy was crucial for diagnosing both celiac disease and ulcerative jejunitis in this pediatric patient.
- The patient was diagnosed with celiac disease and ulcerative jejunitis, confirmed by push enteroscopy and biopsies.
- A strict gluten-free diet resulted in complete endoscopic and histological normalization of the small bowel within one year.
Implications:
- Ulcerative jejunitis in pediatric celiac disease is described for the first time.
- Capsule endoscopy is a valuable tool for diagnosing celiac disease and associated ulcerative jejunitis in children.
- This case suggests that ulcerative jejunitis in pediatric celiac disease may not always indicate refractory disease and can have a positive outcome with dietary management.
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.
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