Chylothorax is a rare clinical condition characterized by high triglyceride and low cholesterol levels in milky pleural aspirate. Generally, it occurs through leakage of chyle as result of trauma or malignancy. Chylothorax due to tuberculous lymphadenitis is very rare clinical condition that has only been documented in a few cases. Although precise pathogenesis is not known, enlarged mediastinal and hilar lymph nodes are thought to be associated with opening of collateral anastomosis between thoracic duct and the azygos and intercostal veins by creating pressure on thoracic duct and cisterna chyli. Presently described is case of chylothorax thought to be due to compression from mediastinal tuberculous lymphadenitis, and which had complete remission after antituberculosis treatment.