Orofacial granulomatosis associated with Crohn's disease

Abstract

Orofacial granulomatosis (OFG) is a term used to describe swelling of the orofacial area, mainly in the lips, secondary to an underlying granulomatous inflammatory process. OFG has been reported in association with systemic conditions such as sarcoidosis and Crohn's disease (CD). OFG may precede gastrointestinal disease, such as CD, by several years and may be the only obvious focus of the disease. Herein, we report a patient with OFG and non-symptomatic ulcerations of the ileocecal valve. The patient received intralesional triamcinolone injections every 2 weeks. After 6 weeks, all oral lesions showed marked improvement. The favourable treatment response of this patient suggests that intralesional triamcinolone can be used as a treatment option for patients with CD that have oral lesions. In addition, patients presenting with OFG should be carefully evaluated for gastrointestinal signs and symptoms.

Keywords: Crohn's disease; Orofacial graulomatosis; Treatment.

Fig. 1

(A) Marked swelling of upper and lower lips. (B, C) Cobble-stone like appearance of the buccal mucosa and sublingual space (asterisk=the site of biopsy). (D) Six weeks after intralesional triamcinolone injections every 2 weeks, the oral lesions showed marked improvement.

Fig. 2

(A, B) Biopsy samples from the buccal mucosa showed non-caseating granuloma with scattered perivascular chronic inflammatory cells (A: H&E, ×100, B: H&E, ×400). (C) The colonoscopic findings showed minor ulceration around the ileocecal valve (black arrow). (D) Mucosal biopsies from the terminal ileum revealed aphthous ulcerations and infiltration by inflammatory cells (H&E, ×400).