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A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
BACKGROUND: Gorham-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 PRESENTATION: A 14-year-old patient presented wi...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2019
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6454674/ https://www.ncbi.nlm.nih.gov/pubmed/30961601 http://dx.doi.org/10.1186/s12891-019-2542-z |
Sumario: | BACKGROUND: Gorham-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 PRESENTATION: A 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. CONCLUSIONS: GSS 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. |
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