Thyroglossal duct cyst is a frequent event; however, papillary carcinoma within a thyroglossal duct cyst is rare, particularly in children. A 17-year-old girl presented with an asymptomatic mid-submental mass for the last 2 months. The diagnosis of thyroglossal duct cyst was made based on physical examination and computed tomography finding. After performance of Sistrunk procedure, an incidental papillary carcinoma within the thyroglossal duct cyst was observed on pathology. We reviewed the pediatric cases of thyroglossal duct carcinoma, and then decided not to perform a concurrent thyroidectomy. We will continue close follow-up for future thyroid involvement.
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