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Gorham-Stout disease of the malleolus: a rare case report
BACKGROUND: Gorham-Stout disease, also known as vanishing bone disease, idiopathic massive osteolysis, is a rare entity of unknown etiopathology. This disease is characterized by destruction of osseous matrix and proliferation of lymphatic vascular structures and associated with massive regional ost...
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/PMC6937978/ https://www.ncbi.nlm.nih.gov/pubmed/31892358 http://dx.doi.org/10.1186/s12891-019-3027-9 |
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author | Zheng, Chuanxi Tang, Fan Min, Li Zhou, Yong Luo, Yi Tu, Chongqi Zhang, Shiquan |
author_facet | Zheng, Chuanxi Tang, Fan Min, Li Zhou, Yong Luo, Yi Tu, Chongqi Zhang, Shiquan |
author_sort | Zheng, Chuanxi |
collection | PubMed |
description | BACKGROUND: Gorham-Stout disease, also known as vanishing bone disease, idiopathic massive osteolysis, is a rare entity of unknown etiopathology. This disease is characterized by destruction of osseous matrix and proliferation of lymphatic vascular structures and associated with massive regional osteolysis. It has a variable clinical presentation and is commonly considered as a benign disease with a progressive tendency and an unpredictable prognosis. The diagnosis is made by exclusion and based on combination with histological, radiological, and clinical features. Despite that several therapeutic options have shown certain efficacy, the effective treatment still remains controversial and there is no standard treatment to be recommended. CASE PRESENTATION: A previously healthy 40-year-old man presented with right lateral malleolus pain after an ankle sprain and was referred to our hospital. The radiographs indicated rapid massive bone destruction in the distal right lateral malleolus with an unclear margin. Based on the combination with histological, radiological, and clinical features, the diagnosis of Gorham-Stout disease was made. Considering that the residual function of malleolus had to be protected, prior bisphosphonate was used to control the progression of lesion, followed by surgical resection and biological reconstruction with autologous fibular bone grafting. The patient was followed up 8 years after surgery, he presented without progression and recurrence. CONCLUSIONS: We depict a case of Gorham-Stout disease at the right lateral malleolus and was successfully controlled by medication and surgical intervention. Based on the prior effective medical treatment, resection with biological reconstruction is a useful approach to treat Graham-Stout disease in bone. |
format | Online Article Text |
id | pubmed-6937978 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-69379782019-12-31 Gorham-Stout disease of the malleolus: a rare case report Zheng, Chuanxi Tang, Fan Min, Li Zhou, Yong Luo, Yi Tu, Chongqi Zhang, Shiquan BMC Musculoskelet Disord Case Report BACKGROUND: Gorham-Stout disease, also known as vanishing bone disease, idiopathic massive osteolysis, is a rare entity of unknown etiopathology. This disease is characterized by destruction of osseous matrix and proliferation of lymphatic vascular structures and associated with massive regional osteolysis. It has a variable clinical presentation and is commonly considered as a benign disease with a progressive tendency and an unpredictable prognosis. The diagnosis is made by exclusion and based on combination with histological, radiological, and clinical features. Despite that several therapeutic options have shown certain efficacy, the effective treatment still remains controversial and there is no standard treatment to be recommended. CASE PRESENTATION: A previously healthy 40-year-old man presented with right lateral malleolus pain after an ankle sprain and was referred to our hospital. The radiographs indicated rapid massive bone destruction in the distal right lateral malleolus with an unclear margin. Based on the combination with histological, radiological, and clinical features, the diagnosis of Gorham-Stout disease was made. Considering that the residual function of malleolus had to be protected, prior bisphosphonate was used to control the progression of lesion, followed by surgical resection and biological reconstruction with autologous fibular bone grafting. The patient was followed up 8 years after surgery, he presented without progression and recurrence. CONCLUSIONS: We depict a case of Gorham-Stout disease at the right lateral malleolus and was successfully controlled by medication and surgical intervention. Based on the prior effective medical treatment, resection with biological reconstruction is a useful approach to treat Graham-Stout disease in bone. BioMed Central 2019-12-31 /pmc/articles/PMC6937978/ /pubmed/31892358 http://dx.doi.org/10.1186/s12891-019-3027-9 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 Zheng, Chuanxi Tang, Fan Min, Li Zhou, Yong Luo, Yi Tu, Chongqi Zhang, Shiquan Gorham-Stout disease of the malleolus: a rare case report |
title | Gorham-Stout disease of the malleolus: a rare case report |
title_full | Gorham-Stout disease of the malleolus: a rare case report |
title_fullStr | Gorham-Stout disease of the malleolus: a rare case report |
title_full_unstemmed | Gorham-Stout disease of the malleolus: a rare case report |
title_short | Gorham-Stout disease of the malleolus: a rare case report |
title_sort | gorham-stout disease of the malleolus: a rare case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6937978/ https://www.ncbi.nlm.nih.gov/pubmed/31892358 http://dx.doi.org/10.1186/s12891-019-3027-9 |
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