Synovial sarcoma represents 5.6%-10% of all soft-tissue sarcomas. Adolescents and young adults are most frequently affected, mainly in the deep soft tissue of the extremities. Only 10% of synovial sarcomas affect the head and neck region; most of these are biphasic. We describe a case of an 18-year-old man who complained of a mass in the right submandibular region that had been present for approximately 12 months. On surgical removal, microscopic analysis showed a tumor formed by sheets of malignant spindle cells involving the submandibular gland. Immunohistochemistry displayed positivity for AE1/AE3, CK18/8, epithelial membrane antigen, CD99, CD56, and TLE-1. Based on these immunohistochemical and histopathologic features, a diagnosis of monophasic synovial sarcoma was rendered. The patient was treated with adjuvant radiotherapy and after 1 year was free of disease. To the best of our knowledge, this is the first reported case of synovial sarcoma involving the submandibular gland. (Oral Surg Oral Med Oral Pathol Oral Radiol 2012;114:e61-e65)1141E61E65