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Serous membrane involvement, including pericardium, peritoneum or pleura, may be observed in more than half of the patients with systemic lupus erythematosus (SLE). However, chylothorax, that is, accumulation of chyle in the pleural space due to SLE is exceedingly rare. The exact pathogenesis of chylothorax in SLE is elusive. SLE-derived inflammation of lymphatic vessels leading to endoluminal back pressure and increased permeability has been hypothesised as the mechanism of development of chylothorax. Due to the rarity of the condition, the ideal treatment protocol is unknown. We hereby present a case of SLE chylothorax with its management and 12-month follow-up. Initially, the patient responded well to immunosuppressive therapy with documented resolution of chylothorax; however, serositis recurred after tapering of steroids, which responded to rituximab.