Two cases of papillary adenocarcinoma originating in the thyroglossal duct remnants are presented. This lesion is a relatively rare one, occurring is less than 1% of benign thyroglossal duct abnomalities. In the literature, 89 cases have been reported, with papillary adenocarcinoma accounting for the vast majority(75-85%) of the lesions. Therapy should be indivisualized, based on the pathologic findings with a combination of surgical and medical procedures. Distant metastasis was very rare and the long term prognosis was very excellent.