The recurrence rate for thyroglossal duct cysts after a Sistrunk operation is 5% compared with 20% if the hyoid cartilage is not removed. However, few guidelines exist when a lesion recurs after an adequate Sistrunk operation. A 2-year-old boy was referred for recurrent thyroglossal duct cyst after complete and adequate resection. Reexploration of the wound and wider excision of the midline cervical tissues failed to treat the problem. Using a combined transoral/cervical approach, a core of tongue around the foramen cecum was removed. A cystic structure was found at pathologic examination. The child remains asymptomatic 24 months later. If thyroglossal duct cysts recur despite an adequate Sistrunk operation, an intralingual remnant should be suspected. Transoral excision of tongue tissue around the foramen cecum may offer a cure.