While pulmonary tuberculosis is a very frequent infection, chylothorax is an unusual manifestation of tuberculous disease. A 71-year-old woman with chylothorax is herein presented. The ductus thoracicus was ligated and lymphadenomegaly, which was adjacent to diaphragm, was resected. Based on the findings of various tests, a diagnosis of tuberculosis lymphadenitis was made. The patient was administered antituberculosis chemotherapy and has since remained asymptomatic for 1 year after the operation. In conclusion, tuberculosis lymphadenomegaly may lead to the development of chylothorax. The combination of appropriate surgical and medical treatment is an effective therapeutic strategy for this pathology.