Chylothorax, a relatively rare condition of pleural effusion, is defined as an accumulation of chyle resulting from damage to the thoracic duct associated with chyle leakage from the lymphatic system into the pleural cavity. The etiologies of this condition are numerous, and the most important approach for management of chylothorax is to determine the definite cause. We report on a case of an unusual complication of chylothorax that occurred after central catheterization and direct instillation of total parenteral nutrition into the pleural cavity.