PURPOSE: To describe 2 patients with orbital invasion by maxillary ameloblastoma, a rare odontogenic tumor that is not commonly encountered in ophthalmic practice. DESIGN: Retrospective, interventional case report. METHODS: Two patients who were diagnosed with maxillary ameloblastoma several years ago sought treatment for new-onset ocular and orbital signs and symptoms. MAIN OUTCOME MEASURES: Clinical and radiological findings and outcome. RESULTS: In the first patient, tumor recurrence with orbital invasion was diagnosed, and the patient underwent a total orbital exenteration. No recurrence was noted after 18 months of follow-up. The second patient had intracranial involvement with orbital invasion and underwent an extensive resection through an intracranial approach. No recurrence was noted after a 6-month follow-up period. CONCLUSIONS: Although a slow-growing tumor, maxillary ameloblastoma can recur after surgical excision and can be locally aggressive; it can invade the orbit and result in significant ocular morbidity. Ophthalmologists should be aware of this tumor and should monitor these patients closely when orbital invasion is suspected.