Extremely rare maxillary unicystic ameloblastoma causing double vision: First case report in the Middle East

Unicystic ameloblastomas are less aggressive than solid (multicystic) variant, unfortunately this subtypes mimics clinically and radiographically more gentle lesions: like odontogenic cysts, so are misdiagnosed unless a histological study is carried out. Moreover, it is clinically silent and normally discovered accidentally. 60-year-old male patient, suffering from a pain and swelling located in the left maxillary region; the patient came with double vision and that was the main compliment. Radiographs showed uniocular radiolucent lesion in the left sinus that contained an impacted third molar. Patient asked for minimal aggressive surgical intervention, and a curettage was preformed along with third impacted molar extraction. Final diagnosis was obtained from histological study, the result was intraluminal unicystic ameloblastoma plexiform subtype. Healing went eventually, patient recovered double vision in one month period and six years follow up showed no recurrence. Unicystic ameloblastoma is a rare odontogenic lesion, with clinical, radiographic and gross features of jaw cysts. The lesion histologically shows typical ameloblastomatous epithelium lining part of the cyst cavity with or without and/or mural tumor growth. Unicystic ameloblastoma usually presents in posterior mandibular ramus region, while it is rare and atypical in posterior maxillary region. Unicystic ameloblastomas with orbital invasion cases are 4 worldwide and this case reports represents the first case in the Middle East. Thorough examination is recommended when unilocular radiolucency of the jaw is detected. Likewise, orbital surgeons are highly advised to take into account the biological behaviors of maxillary odontogenic tumors. •Maxillary unicystic ameloblastoma is a rare case.•Orbital involvement is extremely rare.•Accurate diagnosis, treatment plan and specialties cooperation are important to achieve recovery.•Radiographic and clinical appearance may cause misdiagnosis and later a treatment failure or recurrence.