IntroductionAmeloblastomas are rare head and neck tumors, and yet the most common odontogenic neoplasms. They account for 1% and 11% of all head and neck and odontogenic tumors respectively. Embryologically, they originate from remnants of odontogenic epithelium. Their aggressive, destructive nature, as well as their anticipated high rate of recurrence, even after en bloc resection, poses a surgical predicament.PresentationWe present a case of a 56 year-old Asian female with a multi-recurrent invasive ameloblastoma. Initially, the lesion was mandibular in location for which she underwent a mandiblectomy. Later on, she presented with a maxillary ameloblastoma with invasion of both the anterior wall of the maxillary sinus and the floor of the orbit. The patient was operated twice and histopathology confirmed a cystic type recurrent ameloblastoma. A year later, she came with recurrent maxillary ameloblastoma and a maxillectomy was done. However, histopathology revealed a follicular ameloblastoma. Three years later, she presented with a retro-orbital ameloblastoma with infiltration to the temporal muscles. The patient was operated and the histopathologic examination revealed a partially cystic lesion with no malignant transformation.ConclusionThis case discusses available treatment options and emphasizes on the importance of long-term patient follow-up due to the biological behavior of ameloblastoma.