Ameloblastoma (AM) is a rare epithelium-derived odontogenic tumor, mostly involving the mandible and less often the maxilla. Most AMs are benign and characterized by indolence and local invasiveness, with a high recurrence rate. Herein, we present a case of maxillary AM in a 42-year-old female suffering from left nasal congestion and facial swelling for almost one month after endoscopic surgery at a local hospital. The mass was completely resected by a transnasal functional endoscopic sinus surgery based on radiographic examination. Subsequently, postsurgical histopathological examinations were conducted, and she was diagnosed with a plexiform AM pattern. Immunohistochemical staining revealed that the tumor was positive for PCK, P63, CK5/6, and CK14 but negative for S100, ER, and Ki67. Based on these findings, the patient was diagnosed with maxillary AM.