We describe an extremely rare case of bilateral intrathyroidal branchial cleft cysts. A 2-day-old male neonate presented with intermittent tachypnea, dyspnea, and a palpable neck mass. Cervical ultrasonography and computerized tomography scan demonstrated a right-sided 3 × 3-cm cystic mass and a left sided 4.5 × 4-cm cystic mass, thought to be originating in the thyroid gland. The right-sided cyst decreased in size after OK-432 sclerotherapy. The left-sided cyst showed little response to 2 attempts...