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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...

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Autores principales: Zhang, Hanwen, Han, Chaofan, Pang, Daming, Yong, Hai, Yang, Jincai, Yin, Peng, Zhou, Lijin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
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.
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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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