Schwannomas are benign peripheral nerve sheath tumors and rarely arise in the infratemporal fossa. There are only 10 cases reported in the English literature. The most common clinical manifestations are cheek swelling, facial pain, proptosis and tonsillar fossa swelling. We encountered a 23-year-old female who presented with progressive painless right cheek swelling for one year. Physical examinations revealed a smooth firm mass lateral to the right maxillary molars. CT and MRI revealed a solitary, well-circumscribed soft tissue tumor located behind the posterior wall of the right maxillary sinus. Provisional diagnosis based on the images study suspect a neurogenic tumor. Schwannoma was comfirmed by transoral biopsy. The patient underwent tumor excision via transoral approach. No evidence of recurrence or neurologic deficits is noted after 2 years. Transoral approach provides an alternative way to remove benign tumor limited in the infratemporal fossa.