BACKGROUND Schwannoma is a benign, solitary, and slowly progressive encapsulated tumor originating from the sheath of myelinated nerve fibers. Schwannoma of the salivary gland is a particularly rare form of an extracranial neurogenic tumor. Here, we present an unusual case of a schwannoma of the submandibular gland in a 19-year-old man. Total excision of the submandibular gland resulted in complete resolution of symptoms with no cranial nerve deficits. The details of the histopathologic and immunohistochemical features are presented. METHODS A 19-year-old Caucasian man was admitted to our clinic with a painless mass on the right side of his neck that he had had for the past four months. A neck examination revealed a smooth-surfaced, mobile, firm, and painless mass, 6cm in its greatest diameter, on the right side of the submandibular region. Fine-needle aspiration cytology was suggestive of a submandibular gland schwannoma. After the initial evaluation, our patient was prepared for surgical evaluation and resection with a presumptive diagnosis of a neurogenic tumor of the submandibular gland. The final diagnosis of schwannoma was verified by microscopic and immunohistochemical studies. At one-year follow-up of the case, there was no evidence of recurrence. CONCLUSIONS Schwannoma of the salivary gland is a particularly rare form of an extracranial neurogenic tumor. Our findings indicate good prognosis in an unusual case of a submandibular gland schwannoma in a 19-year-old man treated by surgical excision with no recurrence within 12 months of follow-up.