Abstract: This paper described a patient who suffered from ameloblastoma in the sublingual gland. Clinical examination found an oval-shaped mass in the left floor of the mouth. Imaging examination revealed that the mass located in the left sublingual gland area and did not invade the mandible. Preoperative examinations confirmed that there were no surgical contraindications, then surgical removal of the tumor was performed, and there was no evidence of recurrence 5 months after surgery. Histological examination showed that the tumor was closely related to the sublingual gland, the final pathologic diagnosis was peripheral ameloblastoma.

Key words: Sublingual gland, Peripheral ameloblastoma, Extragingival peripheral ameloblastoma