Thyroglossal duct cysts are one of the most common congenital cervical pathologic findings in children. This type of cyst can be located anywhere between the base of the tongue and the sternal manubrium. We report the case of a patient with a double thyroglossal cyst, one located in the hyoid region and the other in the thyroid gland. The 2 cysts were connected by a permeable tract, which confirms that an involution failure of the embryonic remains of the thyroglossal duct was responsible for the development of the cyst. Intrathyroid cysts of the thyroglossal duct, though uncommon, should be included in the differential diagnosis of thyroid cysts in children.
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