Papillary carcinoma originating from a thyroglossal cyst represents an infrequent finding, which occurs in ~1% of cases of thyroglossal cyst and its presentation is usually the same as that of a benign cyst, which represents the most frequent benign congenital lesion of the neck. The diagnosis is usually presented as a finding on the histopathological examination. Two cases of a thyroglossal cyst with later development of malignancy are presented. The first, a 30-year-old female, and the second, a 32-year-old male. Both began their condition after presenting an increase in volume in the neck, the female presented a right sub-mental triangle of 1 year of evolution, and the male on the anterior side of the neck. In both cases, the diagnosis of thyroglossal cyst was made by the physical examination and findings of cervical ultrasound and computed axial tomography. Surgical management consisted of the Sistrunk procedure with a definitive histopathological result for papillary thyroid carcinoma originating from thyroglossal cyst. In the case of the male patient, the Sistrunk procedure was performed along a total thyroidectomy, although in the female patient, a total thyroidectomy was performed in a second stage. Patients were then discharged and referred to the endocrinology service for further medical treatment. The management of these cases continues to be controversial due to the limited number of reported cases, so the multidisciplinary management and individualization of each case plays a fundamental role in the management of this rare condition.

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