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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 |
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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. |
format | Online Article Text |
id | pubmed-6454674 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
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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