Oral Surg Oral Med Oral Pathol Oral Radiol. 2026 Feb 25:S2212-4403(26)00091-X. doi: 10.1016/j.oooo.2026.02.016. Online ahead of print.
ABSTRACT
INTRODUCTION: Orofacial granulomatosis (OFG) is a rare inflammatory disorder of the oral and perioral tissues that may occur in isolation or as an early manifestation of Crohn’s disease (CD). Its variable presentation often delays recognition of the underlying systemic disease.
CASE REPORT: An 8-year-old boy presented with persistent perioral erythema, lip swelling, cobblestone-like gingival changes, and gastrointestinal symptoms, including constipation and perianal discomfort. Initial differential diagnoses included OFG and angioedema. Early lab tests and labial mucosa biopsy were nonspecific, delaying diagnosis. Repeat biopsies of mandibular gingiva and buccal mucosa revealed noncaseating granulomatous inflammation, confirming OFG. Elevated anti-Saccharomyces cerevisiae antibodies (ASCA IgG) prompted endoscopy, which confirmed CD 9 months after initial oral symptom onset. Systemic therapy with infliximab led to rapid and substantial improvement of oral lesions, perioral swelling, and functional impairment.
CONCLUSION: This case highlights that OFG can represent an early presentation of CD, emphasizing the need for careful biopsy, longitudinal follow-up, and multidisciplinary evaluation. It also demonstrates the effectiveness of biologic therapy with infliximab in managing severe OFG and preventing long-term orofacial complications.
PMID:41862370 | DOI:10.1016/j.oooo.2026.02.016
