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Ectomesenchymal chondromyxoid tumor (ECMT) of the oral cavity is a rare lesion. We describe the 33rd reported case in the current English-language literature. This patient had originally presented 5 years earlier with a tongue neoplasm with biopsy results consistent with nerve sheath myxoma. A general surgical pathologist gave the patient's current lesion a preliminary diagnosis of low-grade sarcoma. After further evaluation by an oral and maxillofacial pathologist, the diagnosis of ectomesenchymal chondromyxoid tumor was established. It is important for clinicians to have a better understanding of ECMT, so as to avoid future misdiagnosis of this entity.