Thyroglossal duct anomalies are the most common malformations in the neck and constitute 70% of all the congenital cervical masses. They are more common in the pediatric population under 5 years of age, and 60% of lesions are diagnosed before the age of 20. They represent remnants from the embryological migration of thyroid tissue from foramen caecum to the thyroid fossa. The thyroglossal duct cyst may rupture spontaneously and present as a draining sinus, which has been erroneously called a thyroglossal fistula, although communication with foramen caecum is extremely rare. We hereby present a case of true thyroglossal fistula in a 21-year-old male with a fistulous communication between the neck skin and foramen caecum.
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