We report the first two published cases of a facial localization of Mycobacterium ulcerans ulcer (Buruli ulcer). M. ulcerans is the third most common mycobacteriosis, after leprosy and tuberculosis. Clinical manifestations involve large necrotic ulcerations with protruding edges, mainly localized on the limbs. Both of our patients, 9 and 45-year-old females, resided in endemic areas of Buruli ulcer in the Ivory Coast. The ulcers were located on the left zygomatic malar region in one patient and the submental region in the other. Treatment involved resection of the ulcer followed by total skin graft. Ulcer healing was observed after two months hospitalization in one patient. The other patient died.