We describe a case of chylothorax in a female patient with a large retrosternal goiter. After subtotal thyroidectomy by means of a Kocher cervicotomy, the chylothorax disappeared in 48 h. At present, 2 years after surgical removal, the patient remains asymptomatic, without recurrent pleural chylous effusion. Until now, to our knowledge, only one such case had been reported.