The thyroglossal duct cyst is the neck congenital abnormality most common in the childhood. Clinically, it shows itself as a solitary neck mass in the region of hyoid bone, it is painless and can be dislocated during de physical examination and tongue protrusion. The most common treatment is its removal through the Sistrunk technique. Patient with diagnosis of thyroglossal duct cyst in neck region with extension to the mouth floor was treated by total surgical removal through intraoral access. The intraoral approach to the treatment of the thyroglossal duct cyst showed itself practicable and permitted the total removal of the lesion with no recurrence signs.