From Orofacial Granulomatosis to Crohn's Disease: A Case Report.

INTRODUCTION: Crohn's disease (CD) is a chronic inflammatory disorder that may involve any segment of the gastrointestinal tract, including the oral cavity. Oral granulomatous inflammation can represent the earliest manifestation of CD or be provisionally classified as orofacial granulomatosis (OFG). Differentiating isolated OFG from early oral CD is challenging, as the intestinal involvement may be subclinical or develop later, and oral clinical and histological findings are not specific. CASE PRESENTATION: We report the case of a 26-year-old woman with biopsy-proven oral granulomatous inflammation initially diagnosed as OFG only partially responsive to topical corticosteroids. Five years later, the patient developed progressive dysphagia, bloody diarrhea, and concomitant draining perianal lesion. Investigations revealed segmental granulomatous colitis, intersphincteric fistula, and a regular nonspecific esophageal stenosis. Colonic CD with oral, esophageal, and perianal involvement was diagnosed. Treatment with corticosteroids followed by infliximab and azathioprine achieved sustained clinical and endoscopic colonic remission with marked improvement of oral, esophageal, and perianal disease. CONCLUSION: This case emphasizes that granulomatous oral inflammation may represent the earliest manifestation of CD and highlights the importance of systematic gastrointestinal evaluation and long-term surveillance even in the absence of digestive symptoms.