We report the case of a 32-year old woman with an anterior cervical cystic mass originating in the thyroglossal duct. At surgical exploration, the mass was reacted, including the thyroglossal duct and the mid-portion of the hyoid bone (Sistrunk's procedure). The histopathologic study demonstrated a papillary carcinoma. Fine needle aspiration biopsies of both thyroid lobes were performed postoperatively without any histopathologic abnormalities. The patient was discharged without performing any other surgical procedure and without complications. Treatment with I131 and thyroid hormones was initiated, and at 11 months of follow-up, the patient is asymptomatic without any evidence of local or distant recurrence. The clinical and histopathological features are discussed, as well as the therapeutic options for this type of uncommon malignant neoplasm.