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The thyroglossal duct cyst, mainly appearing as a midline neck swelling in adolescence and young adults, is the most comon protruding the tongue. Carcinoma arising in thyroglossal duct cyst is found in approximately 1% of these lesions, therefore the presentation of the patient with carcinoma is indistinguishable from the comon cyst. A 50-year-old man was examined preope-ratively by ultrasonography of the neck, asay of thyroid hormones and neck CT with enhancement, which yielded a diagnosis of a thyroglossal duct cyst. The patient underwent Sistrunk procedure for excision of a thyroglossal duct cyst. No thyroid abnorma-bilateral neck dissection 17 days after the Sistrunk procedure. There were no problems in the postoperative course and no signs of local recurrence or metastases through one year after surgery. We present this case of papillary carcinoma arising in the thyroglo-ssal duct cyst, with the review of literature. (Korean J Otolaryngol 2003 ;46 :448-50)