Thyroglossal duct cysts (TGDCs) are usually midline structure of the neck. The coexistence of carcinomas in TGDCs is found rarely, with most being papillary carcinomas. Usually, the diagnosis is made postoperatively after excision of the cyst. Although the Sistrunk procedure is often regarded as adequate, various controversies exist concerning the need for thyroidectomy depending on histopathological findings. We are reporting the case of a 56-year-old man, diagnosed with papillary carcinoma within a TGDC, who underwent total thyroidectomy as has been recommended for differentiated papillary cancer.