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Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review

BACKGROUND: The Gorham–Stout syndrome (GSS), also known as phantom bone disease, is a rare bone condition of unknown etiology. Involvement of the spine is described in <50 cases in the literature. Here, we report a case of thoracic spine fracture dislocation in a young female who was known to hav...

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Autores principales: Jaccard, Alexandre, Macedo, César, Castro, Gabriel, Guiroy, Alfredo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6238329/
https://www.ncbi.nlm.nih.gov/pubmed/30533270
http://dx.doi.org/10.4103/sni.sni_311_18
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author Jaccard, Alexandre
Macedo, César
Castro, Gabriel
Guiroy, Alfredo
author_facet Jaccard, Alexandre
Macedo, César
Castro, Gabriel
Guiroy, Alfredo
author_sort Jaccard, Alexandre
collection PubMed
description BACKGROUND: The Gorham–Stout syndrome (GSS), also known as phantom bone disease, is a rare bone condition of unknown etiology. Involvement of the spine is described in <50 cases in the literature. Here, we report a case of thoracic spine fracture dislocation in a young female who was known to have GSS. CASE DESCRIPTION: A 23-year-old female developed a left spontaneous hemothorax 10 years previously along with left ribs lytic lessions. Pleural and rib biopsies diagnosed angiomatosis and the clinical diagnosis of GSS was established. Following a minor trauma, she presented with a Frankel B deficit attributed to a T3-T4 fracture dislocation with severe spinal cord compression. The patient underwent halo traction with CT. Following reduction, decompression and C5 to T8 with instrumented fusion (posterior only), she neurologically improved to Frankel D, 2 years postoperatively. CONCLUSION: Although GSS is a rare condition in the spine, it may lead to gross instability and catastrophic vertebral fracture/dislocation with paraparesis. Acute spinal cord decompression with stabilization may be warranted to achieve neurological improvement.
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spelling pubmed-62383292018-12-07 Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review Jaccard, Alexandre Macedo, César Castro, Gabriel Guiroy, Alfredo Surg Neurol Int Spine: Case Report BACKGROUND: The Gorham–Stout syndrome (GSS), also known as phantom bone disease, is a rare bone condition of unknown etiology. Involvement of the spine is described in <50 cases in the literature. Here, we report a case of thoracic spine fracture dislocation in a young female who was known to have GSS. CASE DESCRIPTION: A 23-year-old female developed a left spontaneous hemothorax 10 years previously along with left ribs lytic lessions. Pleural and rib biopsies diagnosed angiomatosis and the clinical diagnosis of GSS was established. Following a minor trauma, she presented with a Frankel B deficit attributed to a T3-T4 fracture dislocation with severe spinal cord compression. The patient underwent halo traction with CT. Following reduction, decompression and C5 to T8 with instrumented fusion (posterior only), she neurologically improved to Frankel D, 2 years postoperatively. CONCLUSION: Although GSS is a rare condition in the spine, it may lead to gross instability and catastrophic vertebral fracture/dislocation with paraparesis. Acute spinal cord decompression with stabilization may be warranted to achieve neurological improvement. Medknow Publications & Media Pvt Ltd 2018-11-01 /pmc/articles/PMC6238329/ /pubmed/30533270 http://dx.doi.org/10.4103/sni.sni_311_18 Text en Copyright: © 2018 Surgical Neurology International http://creativecommons.org/licenses/by-nc-sa/4.0 This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as appropriate credit is given and the new creations are licensed under the identical terms.
spellingShingle Spine: Case Report
Jaccard, Alexandre
Macedo, César
Castro, Gabriel
Guiroy, Alfredo
Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title_full Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title_fullStr Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title_full_unstemmed Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title_short Thoracic spine dislocation in Gorham–Stout Syndrome: Case report and literature review
title_sort thoracic spine dislocation in gorham–stout syndrome: case report and literature review
topic Spine: Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6238329/
https://www.ncbi.nlm.nih.gov/pubmed/30533270
http://dx.doi.org/10.4103/sni.sni_311_18
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