A case of a mandibular acanthomatous ameloblastoma, with an admixed little component of follicular type, is reported. The epidemiological features of the present case agree with those reported in the international literature. Clinico-radiographic differential diagnostic difficulties existed with several other noncystic osteolytic lesions of the mandible. Recurrence was diagnosed early 1 year after the initial excisional biopsy, and a definitive rim resection was therefore performed. No further recurrence occurred in the following 4-year follow-up.