Abstract Background: Ameloblastoma is a rare and aggressive odontogenic tumor, more commonly occurring in the mandible than the maxilla. Its clinical presentation in the maxilla can be subtle, often leading to diagnostic delays. The maxillary location poses additional challenges due to proximity to critical anatomical structures, complicating surgical management and reconstruction. Case Presentation: This case involves a 33-year-old male who presented with unilateral epistaxis, nasal obstruction, rhinorrhea, and a gradually enlarging, painless swelling on the right cheek over 3 months. Physical examination revealed a smooth mass in the right nasal cavity extending laterally, without proptosis or neurological deficits. A contrast-enhanced computed tomography scan of the paranasal sinuses identified a large, enhanced soft tissue mass encroaching the right maxillary antrum with displacing of surrounding bone and invasion into adjacent structures. Histopathological analysis confirmed a conventional type of ameloblastoma with mixed follicular and plexiform patterns, and the biopsy came as margin-free. The patient underwent a multi-step surgical intervention, including functional endoscopic sinus surgery with skull base repair, followed by a Weber-Fergusson approach for total maxillectomy to remove the tumor and repair the affected areas. The unique aspect of this case is the tumor ’ s extension into the nasal ethmoid complex and frontal sinus , an extremely rare occurrence in ameloblastoma progression . Conclusion: Maxillary ameloblastomas present diagnostic and therapeutic challenges due to their proximity to critical anatomical structures. A combination of advanced imaging, precise surgical techniques, and thorough histopathological analysis is crucial for effective management. Ensuring clear surgical margins and diligent follow-up are key to minimizing recurrence and optimizing patient outcomes.
Ghaida Alotaibi (Tue,) studied this question.