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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...

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Autores principales: Wang, Peng, Liao, Wenbo, Cao, Guangru, Jiang, Yongyan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
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
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author Wang, Peng
Liao, Wenbo
Cao, Guangru
Jiang, Yongyan
author_facet Wang, Peng
Liao, Wenbo
Cao, Guangru
Jiang, Yongyan
author_sort Wang, Peng
collection PubMed
description 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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spelling pubmed-64546742019-04-19 A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report Wang, Peng Liao, Wenbo Cao, Guangru Jiang, Yongyan BMC Musculoskelet Disord 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 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. BioMed Central 2019-04-09 /pmc/articles/PMC6454674/ /pubmed/30961601 http://dx.doi.org/10.1186/s12891-019-2542-z Text en © The Author(s). 2019 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Wang, Peng
Liao, Wenbo
Cao, Guangru
Jiang, Yongyan
A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title_full A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title_fullStr A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title_full_unstemmed A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title_short A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
title_sort rare case of gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report
topic Case Report
url 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
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