Granulomatous Upper Gastrointestinal Inflammation in Pediatric Ulcerative Colitis

Abstract

Objectives: Differentiating ulcerative colitis (UC) and Crohn disease (CD) can be clinically challenging, especially in children. Granulomatous inflammation has traditionally been attributed to CD. Crypt-associated giant cells and granulomas, however, have been observed in colonic biopsies of patients with UC. This phenomenon has not been described in the upper gastrointestinal (UGI) tract with UC.

Methods: Seven pediatric patients with UC with granulomatous UGI (gUGI) lesions were identified. Diagnosis of UC was based on symptoms, clinical course, laboratory results, imaging, and endoscopy. We compared the gUGI patients to a large cohort of pediatric patients with UC (n = 149).

Results: All fully evaluated cases were associated with bloody diarrhea and moderate to severe pancolitis. Gastric and/or duodenal biopsies demonstrated giant cells or granulomas near gland destruction. Small bowel imaging did not reveal any involvement. The majority of cases responded to standard medical therapies, except for 2 patients (28.6%) who required total colectomy. Acute severe, refractory colitis (ie, colectomy within 1 month of presentation) was significantly more common in the gUGI group than the large pediatric UC group (28.6% vs 1.3%, Fisher exact P = 0.01).

Conclusions: This is the first report of pediatric UC-associated granulomatous inflammation in the UGI tract. We speculate that these lesions represent extracolonic manifestations of intense colonic disease. These atypical findings expand the diagnostic considerations that should be incorporated during the differentiation between UC and CD in the pediatric age group.

Conflict of interest statement

Conflicts of Interest and Source of Funding: KQ was supported by the Thrasher Early Career Award for work that is unrelated to this manuscript. FDI was supported by grant NIH T32 DK007664-24S1. RK was funded by philanthropic support from the Men of Distinction (Houston, TX) and the Gutsy Kids Fund supported by the Karen and Brock Wagner family. For the remaining authors, no conflicts of interest or sources of funding were declared.

Figures

Figure 1

Endoscopy and pathology in Patient D. Antrum (top row) had superficial erosions and duodenum (bottom row) had mild edema seen endoscopically. Associated biopsies (Hematoxylin-eosin staining) from these locations demonstrated giant cells (see arrow; 40× magnification, top right) and granulomatous inflammation (arrow; 20× magnification, bottom right) near crypt degeneration.