Synovial sarcoma is usually found near large joints and bursae of the extremities. Only 10% of all synovial sarcomas occur in the head and neck. The most common site affected in the head and neck is the hypopharynx, and very rare in the infratemporal fossa. Treatment of synovial sarcoma of the head and neck is difficult and must have multimodality which consists of operation, radiation and chemotherapy. Prognosis is associated with completeness of resection. We reported a case of synovial sarcoma arising from the infratemporal fossa, the first case in Korea, with review of literature.