2084 An Atypical Case of Oral Crohn’s Disease

Abstract

INTRODUCTION: Oral manifestations of Crohn’s disease are wide-ranging from fissuring and linear ulcerations to cobblestone plaques, mucosal tags, and buccal edema. The commonality between them is the presence of subepithelial non-caseating granulomas on biopsy not explained by another etiology. There is general agreement that oral manifestations are underdiagnosed and that there may be a wider spectrum of disease than previously realized. We present a case of oral Crohn’s that differs in presentation from others previously described in the literature. CASE DESCRIPTION/METHODS: 25-year-old woman who was diagnosed with Crohn’s ileocolitis on colonoscopy in 2011. After initial treatment failure with mesalamine, she was on Infliximab for 3-4 years but stopped as she preferred to avoid infusions. She started Humira in January 2017 after a Crohn’s flare but did not improve despite the addition of 6-MP. She stopped both Adalimumab and 6-MP in September 2017 after developing facial and lip swelling concerning for an allergic reaction. Complement and C1 esterase inhibitor levels were within normal limits, ruling out hereditary angioedema. Her abdominal symptoms worsened off treatment; colonoscopy revealed moderately active left-sided colitis. While her facial swelling improved, her lip swelling did not. On exam she initially had cobblestoning, a tongue fissure, and swelling of the lower lip. She was evaluated by oral and maxillofacial surgery (OMFS) in May 2018 and at that time had a firm, mobile 1.5 × 1 cm exophytic lesion with surface ulcerations of the right buccal mucosa and right lower lip swelling. The buccal lesion was thought to be either a traumatic fibroma or oral Crohn’s disease. It was excised a month later and pathology revealed granulomatous inflammation with no organisms on gram stain or AFB stain, consistent with oral Crohn’s disease. She subsequently started Ustekinumab and attained clinical and endoscopic remission. An oral prednisone taper for her lip swelling had no effect. She then had 2 triamcinolone injections by OMFS with improvement in the lip appearance. DISCUSSION: In this case a woman with intestinal Crohn’s disease developed an exophytic lesion of the buccal mucosa diagnosed as oral Crohn’s disease on biopsy. This atypical presentation of an underdiagnosed aspect of Crohn’s disease points to a wider spectrum of the disease than previously acknowledged. It also highlights the importance of an interdisciplinary approach with dentistry to better recognize and treat oral symptoms.