A 33-year-old patient, Known case of chronic kidney disease on maintenance dialysis presented with complaints of low-grade fever and weight loss of 2 months duration. Computed tomography (CT) revealed bilateral mild pleural effusion with significant mediastinal and abdominal adenopathy. CT-guided fine-needle aspiration cytology of abdominal lymph nodes and bone marrow culture was suggestive of tuberculosis. The patient was started on four drug anti-tubercular therapy, post 6 weeks of initiation he developed new onset fever and chest X-ray revealed moderate right pleural effusion. Diagnostic thoracocentesis was suggestive of chylothorax. To the best of our knowledge, this is the first case report of chylothorax due to the paradoxical reaction in the HIV-negative tuberculous patient.