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Gorham-Stout disease: interesting cause of pleural effusion. Gorham-Stout disease (GSD) is a rare disorder characterised by massive painless osteolysis due to lymphangiomatous tissue progression. GSD's pathogenesis is still unclear, but osteoclasts' activation may play a role in its pathogenesis. There are multiple complications associated with GSD. One of the most severe and life-threatening complications is a chylothorax. Herein we discuss a case of a patient with a history of GSD who presented to the hospital with progressive dyspnoea secondary to a large left-sided pleural effusion, which was later confirmed to be a chylothorax. We will further discuss the current literature and treatment of chylothorax associated with GSD.