A 28-year-old man presented with a 2-year history of facial lesions with discharge (Panels A and B) that had developed 6 years after a diagnosis of Crohn’s disease, despite the administration of immunosuppressant and biologic therapies. He had fistulating disease and had previously undergone surgical resections that resulted in an ileostomy and a rectal stump. Magnetic resonance imaging of the orofacial region (Panel C) and radiography performed after the infusion of the sinus tract with radiopaque material revealed three orocutaneous fistulae arising from inflamed oral mucosa associated with dental disease (Panel A). Extraction of the teeth and curettage of the sinus tract revealed granulomatous inflammation with no infective organisms. Crohn’s disease is characterized by chronic intestinal granulomatous inflammation that may affect any part of the gastrointestinal tract, from the mouth to the anus. Granulomatous conditions affecting the orofacial region are rare. Treatment with antibiotics after extraction of the teeth resulted in resolution of the fistulae. At follow-up after 1 year, the patient had no recurrence of orofacial disease and underwent a successful completion proctectomy. He is currently being treated with adalimumab and methotrexate and has had no obvious recurrence of disease.