Papillary carcinoma arising in a submentalintralingual thyroglossal duct cyst

An extremely rare case of papillary carcinoma is described arising in a submental-intralingual thyroglossal duct cyst. A 27-year-old male presented with a painless swelling in the submental region extending to the tongue which had gradually been increasing in size over the last three months. Upon examination of the neck, a tender right paramedian mass was found in the submental region just above the hyoid bone, it was mobile upon swallowing without clinical evidence of cervical nodes. The Sistrunk surgical technique, extended to the submental and intra lingual region was used to radically remove the mass. The occurrence of carcinoma of the thyroglossal duct cyst, even though rare and unexpected, should always be considered an option, and histologically ruled out mainly on account of the atypical localization, such as the submental-intralingual reported herein. The two different surgical approaches reported in the literature, one more conservative and the other more aggressive, apparently alternatives, are, instead, complementary and adequate when strict diagnostic criteria and adequate follow-up, are observed.