Abstract
BackgroundGorham-Stout syndrome (GSS) with spinal involvement is extremely rare, and bilateral chylothorax as a complication is usually fatal. In our case, pleural effusion appeared in the left hemithorax after ligating the right thoracic duct.Case presentationA 14-year-old patient presented with GSS affecting the thoracic spine with bilateral chylothorax. The case was successfully managed using combined conservative and surgical treatments. At the 2-year follow-up visit, the amount of pleural fluid was reduced, the patient’s respiratory function had improved, and the deformity on the thoracic spine had gradually stabilized.ConclusionsGSS is a rare disorder of the musculoskeletal system that responds poorly to therapies and exhibits very high morbidity and mortality. Chylothorax is a common complication when lesions involve the thoracic spine, and physicians should be vigilant for possible serious pulmonary complications.
Highlights
ConclusionsGorham-Stout syndrome (GSS) is a rare disorder of the musculoskeletal system that responds poorly to therapies and exhibits very high morbidity and mortality
Gorham-Stout syndrome (GSS) with spinal involvement is extremely rare, and bilateral chylothorax as a complication is usually fatal
Chylothorax is a common complication when lesions involve the thoracic spine, and physicians should be vigilant for possible serious pulmonary complications
Summary
This report describes a case of GSS affecting the thoracic spine with progressive bilateral chylothorax, which was treated successfully. GSS is a rare disorder of the musculoskeletal system that responds poorly to therapies and has high morbidity and mortality. Chylothorax is a common complication, and physicians should be vigilant about possible serious pulmonary complications when lesions involve the thoracic spine and make appropriate management decisions. A review of the literature as well as the results of the present case support the need for further studies to elucidate the pathogenesis of GSS with chylothorax and seek effective therapies
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