The occurrence of carcinoma of the thyroglossal duct remnant (TDRCa) is reported to be less than 1%. We retrospectively analyzed 18 cases (14 females, 4 males; mean age, 38.17 years) of TDRCa who underwent surgery in our department, to evaluate the correct workup for diagnosis and treatment. Nine patients underwent a total thyroidectomy and a Sistrunk procedure (SP) whereas 8 underwent a completion total thyroidectomy after a postoperative diagnosis of TDRCa. One patient was diagnosed with TDRCa several years after total thyroidectomy for goiter. Postoperatively all patients were given radioiodide and levothyroxine therapy. Mean follow-up was 85 months. No patient died of tumor-related disease. Thyroglossal duct neoplasms were represented by papillary carcinoma in 16 cases; thyroid histology demonstrated papillary carcinoma in 6 cases (33.3%). Three patients demonstrated single regional node metastasis and only one of them demonstrated the presence of malignant thyroid disease. When clinical diagnosis of thyroglossal duct cyst is made, the workup should be completed by ultrasound (US) scan and fine-needle aspiration cytology (FNAC) in order to plan the correct surgery for a possible TDRCa. Total thyroidectomy should always be considered with SP to permit the correct treatment and follow-up in those patients. Long-term survival of patients with TDRCa is excellent.
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