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A report of two children with Gorham-Stout disease

BACKGROUND: Gorham-Stout disease is a rare condition characterized by unifocal and massive type IV osteolysis (variant of idiopathic nonhereditary osteolytic disease) with a slow progression, which is self-limiting for some years. It is characterized by recurrent vascular tumors with disruption of t...

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Autores principales: Tena-Sanabria, Mario Edgar, Jesús-Mejenes, Larisa Yarindy, Fuentes-Herrera, Gabriela, Álvarez-Martínez, Félix Alejandro, Victorio-García, Nora Patricia, Núñez-Enríquez, Juan Carlos
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6591827/
https://www.ncbi.nlm.nih.gov/pubmed/31234820
http://dx.doi.org/10.1186/s12887-019-1561-0
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author Tena-Sanabria, Mario Edgar
Jesús-Mejenes, Larisa Yarindy
Fuentes-Herrera, Gabriela
Álvarez-Martínez, Félix Alejandro
Victorio-García, Nora Patricia
Núñez-Enríquez, Juan Carlos
author_facet Tena-Sanabria, Mario Edgar
Jesús-Mejenes, Larisa Yarindy
Fuentes-Herrera, Gabriela
Álvarez-Martínez, Félix Alejandro
Victorio-García, Nora Patricia
Núñez-Enríquez, Juan Carlos
author_sort Tena-Sanabria, Mario Edgar
collection PubMed
description BACKGROUND: Gorham-Stout disease is a rare condition characterized by unifocal and massive type IV osteolysis (variant of idiopathic nonhereditary osteolytic disease) with a slow progression, which is self-limiting for some years. It is characterized by recurrent vascular tumors with disruption of the anatomical architecture and intraosseous proliferation of vascular channels that leads to the destruction and resorption of the bone matrix. The aim of this study is to present the clinical features of this disease, as well as the importance of prompt diagnosis and treatment, with a review of the reported cases. CASE REPORTS: We describe two cases of Gorham-Stout disease between 2013 and 2017 with surgical interventions, follow-up and results. Case one involves an 11-year-old male with involvement of the left iliac bone, with adequate evolution after a surgical procedure with a lyophilized cadaveric tricortical bone allograft. Case two involves a 6-year-old male with cervical spine C1-C3 repercussion; in the protocol for surgical treatment, he presented with signs of spinal cord compression and died. CONCLUSION: Diagnosis of Gorham-Stout disease is made by exclusion, and its clinical presentation varies widely, from spontaneous remission to a fatal outcome. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s12887-019-1561-0) contains supplementary material, which is available to authorized users.
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spelling pubmed-65918272019-07-08 A report of two children with Gorham-Stout disease Tena-Sanabria, Mario Edgar Jesús-Mejenes, Larisa Yarindy Fuentes-Herrera, Gabriela Álvarez-Martínez, Félix Alejandro Victorio-García, Nora Patricia Núñez-Enríquez, Juan Carlos BMC Pediatr Case Report BACKGROUND: Gorham-Stout disease is a rare condition characterized by unifocal and massive type IV osteolysis (variant of idiopathic nonhereditary osteolytic disease) with a slow progression, which is self-limiting for some years. It is characterized by recurrent vascular tumors with disruption of the anatomical architecture and intraosseous proliferation of vascular channels that leads to the destruction and resorption of the bone matrix. The aim of this study is to present the clinical features of this disease, as well as the importance of prompt diagnosis and treatment, with a review of the reported cases. CASE REPORTS: We describe two cases of Gorham-Stout disease between 2013 and 2017 with surgical interventions, follow-up and results. Case one involves an 11-year-old male with involvement of the left iliac bone, with adequate evolution after a surgical procedure with a lyophilized cadaveric tricortical bone allograft. Case two involves a 6-year-old male with cervical spine C1-C3 repercussion; in the protocol for surgical treatment, he presented with signs of spinal cord compression and died. CONCLUSION: Diagnosis of Gorham-Stout disease is made by exclusion, and its clinical presentation varies widely, from spontaneous remission to a fatal outcome. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s12887-019-1561-0) contains supplementary material, which is available to authorized users. BioMed Central 2019-06-24 /pmc/articles/PMC6591827/ /pubmed/31234820 http://dx.doi.org/10.1186/s12887-019-1561-0 Text en © The Author(s). 2019 Open AccessThis 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
Tena-Sanabria, Mario Edgar
Jesús-Mejenes, Larisa Yarindy
Fuentes-Herrera, Gabriela
Álvarez-Martínez, Félix Alejandro
Victorio-García, Nora Patricia
Núñez-Enríquez, Juan Carlos
A report of two children with Gorham-Stout disease
title A report of two children with Gorham-Stout disease
title_full A report of two children with Gorham-Stout disease
title_fullStr A report of two children with Gorham-Stout disease
title_full_unstemmed A report of two children with Gorham-Stout disease
title_short A report of two children with Gorham-Stout disease
title_sort report of two children with gorham-stout disease
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6591827/
https://www.ncbi.nlm.nih.gov/pubmed/31234820
http://dx.doi.org/10.1186/s12887-019-1561-0
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