Thyroid carcinomas arising from ectopic thyroid tissue are uncommon;most of them arise from thyroid tissue in thyroglossal cysts. A rare case of a 66-year-old woman with papillary thyroid carcinoma arising from median ectopic thyroid tissue lacking a thyroglossal duct remnant is reported. The tumor was resected by Sistrunk's procedure, and the patient's postoperative course was good.