Orofacial manifestations of Crohn's disease can be difficult to diagnose and treat. We report a case in which the orofacial lesions occurred 7 years prior to the diagnosis of underlying inflammatory bowel disease. The patient was refractory to mesalamine and systemic corticosteroids but responded to infliximab, the chimeric monoclonal antibody to tumor necrosis factor (TNF-alpha). A review of the literature of the orofacial granulomatoses is presented as well.