Objective: To present a case of metastatic ameloblastoma in a patient who has undergone repeated surgical resections, to discuss its histogenesis, clinical features, distinctive diagnostic characteristics, and management. Results: A 51-year-old man diagnosed twice with ameloblastoma underwent excision via segmental mandibulectomy 15 years prior to admission. Recurrence of the mass necessitated another excision this time with TMJ disarticulation 5 years prior to admission. He was apparently well until 3 years prior to admission when he noticed another mass growing at his infra-auricular area. One month prior to admission, patient noticed another separate mass growing on his ipsilateral neck area. Surgery was done using Weber-Ferguson approach and histopathological results revealed metastatic ameloblastoma. Upon the last examination 4 months after admission, the patient remains tumor free without signs of recurrence.