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Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review
BACKGROUND: Gorham–Stout syndrome is an uncommon condition with a varied clinical presentation and unclear cause that is characterised by a proliferation of lymphatic capillaries and severe regional osteolysis. Spinal and visceral involvement increases the syndrome's morbidity and mortality rat...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9393415/ https://www.ncbi.nlm.nih.gov/pubmed/36003281 http://dx.doi.org/10.3389/fsurg.2022.981025 |
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author | Zhang, Hanwen Han, Chaofan Pang, Daming Yong, Hai Yang, Jincai Yin, Peng Zhou, Lijin |
author_facet | Zhang, Hanwen Han, Chaofan Pang, Daming Yong, Hai Yang, Jincai Yin, Peng Zhou, Lijin |
author_sort | Zhang, Hanwen |
collection | PubMed |
description | BACKGROUND: Gorham–Stout syndrome is an uncommon condition with a varied clinical presentation and unclear cause that is characterised by a proliferation of lymphatic capillaries and severe regional osteolysis. Spinal and visceral involvement increases the syndrome's morbidity and mortality rates. Here, we report about a male patient with Gorham's disease who developed local kyphosis and neurological disorders due to massive osteolysis. CASE PRESENTATION: A 13-year-old male patient presented with progressive kyphosis and massive osteolysis of the thoracic vertebrae. Halo-pelvic traction and vertebral column resection osteotomy were performed to reconstruct the spine and prevent disease progression. The entire lesion was resected, and an artificial vertebra filled with allograft bone was used to achieve temporary stability. Although the patient presented with chylothorax following surgery, which required thoracic drainage, the patient did achieve a satisfying outcome. CONCLUSIONS: Limited by the number of GSS cases with spinal involvement and chylothorax manifestations, halo-pelvic distraction as a preoperative preparation and vertebral column resection osteotomy provide a novel avenue for managing this disease. |
format | Online Article Text |
id | pubmed-9393415 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-93934152022-08-23 Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review Zhang, Hanwen Han, Chaofan Pang, Daming Yong, Hai Yang, Jincai Yin, Peng Zhou, Lijin Front Surg Surgery BACKGROUND: Gorham–Stout syndrome is an uncommon condition with a varied clinical presentation and unclear cause that is characterised by a proliferation of lymphatic capillaries and severe regional osteolysis. Spinal and visceral involvement increases the syndrome's morbidity and mortality rates. Here, we report about a male patient with Gorham's disease who developed local kyphosis and neurological disorders due to massive osteolysis. CASE PRESENTATION: A 13-year-old male patient presented with progressive kyphosis and massive osteolysis of the thoracic vertebrae. Halo-pelvic traction and vertebral column resection osteotomy were performed to reconstruct the spine and prevent disease progression. The entire lesion was resected, and an artificial vertebra filled with allograft bone was used to achieve temporary stability. Although the patient presented with chylothorax following surgery, which required thoracic drainage, the patient did achieve a satisfying outcome. CONCLUSIONS: Limited by the number of GSS cases with spinal involvement and chylothorax manifestations, halo-pelvic distraction as a preoperative preparation and vertebral column resection osteotomy provide a novel avenue for managing this disease. Frontiers Media S.A. 2022-08-08 /pmc/articles/PMC9393415/ /pubmed/36003281 http://dx.doi.org/10.3389/fsurg.2022.981025 Text en © 2022 Zhang, Han, Pang, Yong, Yang, Yin and Zhou. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) (https://creativecommons.org/licenses/by/4.0/) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms. |
spellingShingle | Surgery Zhang, Hanwen Han, Chaofan Pang, Daming Yong, Hai Yang, Jincai Yin, Peng Zhou, Lijin Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title | Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title_full | Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title_fullStr | Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title_full_unstemmed | Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title_short | Surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with Gorham–Stout syndrome: A case report and literature review |
title_sort | surgical treatment of severe thoracic kyphosis and neurological deficit in a patient with gorham–stout syndrome: a case report and literature review |
topic | Surgery |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9393415/ https://www.ncbi.nlm.nih.gov/pubmed/36003281 http://dx.doi.org/10.3389/fsurg.2022.981025 |
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