Although a thyroglossal duct cyst is a congenital anomaly, it can also appear in adults. Despite the presence of embryological remnants, it is still unclear why the cyst should suddenly develop later in life. We report a case of a 46-year-old male with an extravasation mucocele arising from a long-standing lingual thyroglossal duct remnant. MRI demonstrated a lingual cystic lesion near the hyoid bone associated with a suprahyoid tract-like structure masquerading as a thyroglossal duct cyst. However, histopathological examination demonstrated a mucocele secondary to a rupture of a thyroglossal duct remnant with numerous intramural heterotopic salivary glands. We propose a new mechanism of an acquired cystic formation of this congenital disease that excessive production of mucus from heterotopic salivary glands and a physical trauma such as swallowing may lead to extravasation of mucus from the thyroglossal duct.
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