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Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report
BACKGROUND: Gorham-Stout disease (GSD) is a rare osteolytic disease with unknown etiology, varied clinical manifestations and unpredictable prognosis. This disease is characterized by progressive massive local osteolysis and resorption caused by intraosseous lymphatic vessel structure and thin-walle...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Frontiers Media S.A.
2023
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9922857/ https://www.ncbi.nlm.nih.gov/pubmed/36793315 http://dx.doi.org/10.3389/fsurg.2023.1078869 |
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author | Zhang, LiQian Li, JianJian Yao, Fei Chen, YiHao Zhang, Shuo Lv, Hao Jing, JueHua |
author_facet | Zhang, LiQian Li, JianJian Yao, Fei Chen, YiHao Zhang, Shuo Lv, Hao Jing, JueHua |
author_sort | Zhang, LiQian |
collection | PubMed |
description | BACKGROUND: Gorham-Stout disease (GSD) is a rare osteolytic disease with unknown etiology, varied clinical manifestations and unpredictable prognosis. This disease is characterized by progressive massive local osteolysis and resorption caused by intraosseous lymphatic vessel structure and thin-walled vascular proliferation. The diagnosis of GSD has not yet formed a uniform standard, but the combination of clinical manifestations, radiological features and unique histopathological examinations and excluding other diseases contribute to early diagnosis. Although medical therapy, radiotherapy and surgical interventions or combinations have been used for the treatment of GSD, there is currently still no recommended standardized treatment regimen. CASE REPORT: This paper presents a case of a previously healthy 70-year-old man presented with a 10-year history of severe right hip pain and progressive walking disorder of the lower limbs. Based on the patient's clear clinical presentation, unique radiological features, and histological findings, a diagnosis of GSD was made with the exclusion of other potential diseases. The patient was treated with bisphosphonates to slow the progression of the disease followed by total hip arthroplasty to help restore walking function. At the 3-year follow-up, the patient returned to normal walking and no recurrence was observed. CONCLUSION: Bisphosphonates combined with total hip arthroplasty may be an effective method for the treatment of severe GSD in the hip joint. |
format | Online Article Text |
id | pubmed-9922857 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-99228572023-02-14 Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report Zhang, LiQian Li, JianJian Yao, Fei Chen, YiHao Zhang, Shuo Lv, Hao Jing, JueHua Front Surg Surgery BACKGROUND: Gorham-Stout disease (GSD) is a rare osteolytic disease with unknown etiology, varied clinical manifestations and unpredictable prognosis. This disease is characterized by progressive massive local osteolysis and resorption caused by intraosseous lymphatic vessel structure and thin-walled vascular proliferation. The diagnosis of GSD has not yet formed a uniform standard, but the combination of clinical manifestations, radiological features and unique histopathological examinations and excluding other diseases contribute to early diagnosis. Although medical therapy, radiotherapy and surgical interventions or combinations have been used for the treatment of GSD, there is currently still no recommended standardized treatment regimen. CASE REPORT: This paper presents a case of a previously healthy 70-year-old man presented with a 10-year history of severe right hip pain and progressive walking disorder of the lower limbs. Based on the patient's clear clinical presentation, unique radiological features, and histological findings, a diagnosis of GSD was made with the exclusion of other potential diseases. The patient was treated with bisphosphonates to slow the progression of the disease followed by total hip arthroplasty to help restore walking function. At the 3-year follow-up, the patient returned to normal walking and no recurrence was observed. CONCLUSION: Bisphosphonates combined with total hip arthroplasty may be an effective method for the treatment of severe GSD in the hip joint. Frontiers Media S.A. 2023-01-30 /pmc/articles/PMC9922857/ /pubmed/36793315 http://dx.doi.org/10.3389/fsurg.2023.1078869 Text en © 2023 Zhang, Li, Yao, Chen, Zhang, Lv and Jing. 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, LiQian Li, JianJian Yao, Fei Chen, YiHao Zhang, Shuo Lv, Hao Jing, JueHua Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title | Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title_full | Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title_fullStr | Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title_full_unstemmed | Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title_short | Treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: A case report |
title_sort | treatment of gorham-stout disease with bisphosphonates and total hip arthroplasty: a case report |
topic | Surgery |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9922857/ https://www.ncbi.nlm.nih.gov/pubmed/36793315 http://dx.doi.org/10.3389/fsurg.2023.1078869 |
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