Gorham-Stout disease of the mandible, manubrium and cervical spine presenting as bilateral chylothorax.

Gorham-Stout disease (GSD) is an extremely rare musculoskeletal disease of unknown aetiology characterised by non-neoplastic proliferation of vascular and lymphatic channels causing massive osteolysis, typically affecting younger individuals. Chylothorax is a known complication of GSD which is postulated to occur from thoracic spine involvement leading to pleural or thoracic duct invasion. In our case, bilateral chylothorax developed in a 60-year-old woman without any thoracic spine involvement of her disease, challenging the proposed mechanism. Despite bilateral pleural drainage and escalating doses of sirolimus, she ultimately developed respiratory failure and shock and succumbed to her illness. Overall survival of GSD is unknown, but when complicated by chylothorax, prognosis is typically poor. GSD represents a diagnostic and management challenge due to the paucity of knowledge surrounding its aetiology and management. These patients require multidisciplinary coordinated care. It is also important to note its high mortality when associated with chylothorax in particular.

Thompson AA ，Patrawala S 《-》

A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report.

Wang P ，Liao W ，Cao G ，Jiang Y ... -  《BMC MUSCULOSKELETAL DISORDERS》

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.

Olea-Mendoza D ，Terrigno VR ，Balogun A ，Caprio C ... -  《-》

A New Case and Review of Chylothorax in Generalized Lymphatic Anomaly and Gorham-Stout Disease.

Ludwig KF ，Slone T ，Cederberg KB ，Silva AT ，Dellinger M ... -  《LYMPHOLOGY》

Gorham-Stout syndrome with chylothorax in a six-year-old boy.

Deveci M ，Inan N ，Corapçıoğlu F ，Ekingen G ... -  《-》