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Schwannoma, also known as neurilemmoma, is a benign neoplasm that originates from anynerves wrapped with a sheath made of Schwann cells. Schwannoma occurring in the head andneck region is not rare, but schwannomas of the anterior neck, especially ansa cervicalis, areextremely rare that only 7 cases have been reported to date worldwide. Although rare, it shouldbe considered in differential diagnosis of anterior cervical mass and may be confused with othercervical and thyroid mass. We report a case of intramuscular schwannoma in the sternohyoidmuscle. Preoperative diagnosis was established with an ultrasound-guided core needle biopsy. Although it was removed entirely without connection to any other nerves identified or any complication,clinically, the mass was thought to be derived from the nerve. To our knowledge, thisis the first case of the intramuscular schwannoma occurring from ansa cervicalis reported inthe literature.