BACKGROUND Schwannoma is an uncommon benign tumor that develops from Schwann cells in the peripheral nervous system; 25-50% of such tumors occur in the head and neck. A rare variety of schwannoma, known as an ancient schwannoma, exhibits calcification, cystic degeneration, hemorrhage, myxoid stroma, pleomorphism, and nuclear hyperchromatism. Such characteristics may contribute to this tumor being incorrectly diagnosed as malignant. Almost all reported intra-oral ancient schwannomas behave innocuously. CASE REPORT A 20-year-old man presented to the oral surgery clinic with a right facial swelling that began a few months before. CT and MRI were performed and showed a well-defined cystic lesion anterior to the right masseter muscle, located in the subcutaneous area and measuring 28×24×20 mm. No extension to the surrounding tissues nor any other similar lesions were seen. Microscopic examination revealed an encapsulated nodular mass with cystic cavities, hemorrhage, and variably distributed myxoid and hyalinized stroma. The tumor demonstrated 2 distinct cell distributions: hypercellular areas (Antoni A) and myxoid hypocellular areas (Antoni B). Focal areas in high-power magnification showed atypical cells with hyperchromatic and pleomorphic nuclei. The tumor cells showed diffuse positivity for S-100 in IHC. The diagnosis of ancient schwannoma was made. At 1-year follow-up, there was no evidence of nerve injury or recurrence. CONCLUSIONS One more case of oral ancient schwannoma is added to the literature. This is the first reported in Saudi Arabia. It is prudent to be familiar with the degenerative and cytologic alterations of ancient schwannoma, as this may limit the likelihood of misdiagnosis and unnecessary treatment of this benign tumor, with an excellent prognosis.