Ameloblastoma is a benign locally invasive odontogenic tumor. It is the most common odontogenic tumor [1]. Usually, in the middle-aged population, it is more common in the mandible with about 5-20% occurring in the maxilla. We report a case of maxillary unicystic ameloblastoma in a 3.5-year-old girl. Which presented clinically as a large facial swelling causing severe facial deformation and pressure symptoms on the left eye. CT scan showed that the swelling extended to involve the orbital floor. The lesion was diagnosed and confirmed by incisional biopsy then was successfully managed by maxillectomy and immediate reconstruction using the buccal pad of fat. One year follow up with no further complaints and no recurrence was observed. "Written informed consent was obtained from the patient guardians for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request".