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Thyroglossal duct cyst (TGDC) is the most common congenital midline neck mass in children. It usually becomes symptomatic following a respiratory tract infection and is usually diagnosed at 5 years of age. Thyroglossal duct cyst is rarely observed in less than 1-year-old infants. In this study, we present a 3-month-old infant with TGDC, who was administered multiple courses of antibiotic therapy for the hyperemic, draining, midline neck mass that had existed since he was 15 days old. Physical examination revealed an infrahyoid midline neck mass measuring 3× 3 cm, which moved while swallowing and had a sinus opening in the overlying skin. The patient underwent Sistrunk operation under general anesthesia. Histopathologic examination revealed TGDC. One-year follow-up revealed no recurrence.