Two cases of Gorham-Stout disease with good response to zoledronic acid treatment.

Q3 Medicine
María Lorena Brance, Alejandro Castiglioni, Nicolas Cóccaro, Mariano Palatnik
{"title":"Two cases of Gorham-Stout disease with good response to zoledronic acid treatment.","authors":"María Lorena Brance,&nbsp;Alejandro Castiglioni,&nbsp;Nicolas Cóccaro,&nbsp;Mariano Palatnik","doi":"10.11138/ccmbm/2017.14.2.250","DOIUrl":null,"url":null,"abstract":"<p><p>Gorham-Stout disease - also called vanishing bone syndrome - is a rare bone disease characterized by a progressive intra-osseous proliferation of non-neoplastic vascular tissue resulting in massive osteolysis. Here, we report two clinical cases of Gorham-Stout disease. Case 1: a 56-year-old woman with 20 years of history of pain and swell in elbows, ankles and wrist. Then she was diagnosed as systemic lupus erythematosus (SLE) with glomerulonephritis type III. After other pathologies were ruled out Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and after third infusion a progressive improvement of pain, mobility and daily activities were observed. Case 2: a 70-years-old man with a history of pain and limited motion in the left shoulder without X-ray abnormality. Six months later pathological fracture in the left humerus occurred and after ruled out other pathologies Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and a good response was observed after the first infusion. Nowadays just over 200 cases were reported. Gorham-Stout disease was reported in different bones, at different age presentation and severe physical deformities, disabilities, and life-threatening complications can occur. Two cases of Gorham-Stout disease with good response to zoledronic acid was reported in this article.</p>","PeriodicalId":47230,"journal":{"name":"Clinical Cases in Mineral and Bone Metabolism","volume":"14 2","pages":"250-253"},"PeriodicalIF":0.0000,"publicationDate":"2017-05-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5726221/pdf/250-253.pdf","citationCount":"12","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Clinical Cases in Mineral and Bone Metabolism","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.11138/ccmbm/2017.14.2.250","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2017/10/25 0:00:00","PubModel":"Epub","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
引用次数: 12

Abstract

Gorham-Stout disease - also called vanishing bone syndrome - is a rare bone disease characterized by a progressive intra-osseous proliferation of non-neoplastic vascular tissue resulting in massive osteolysis. Here, we report two clinical cases of Gorham-Stout disease. Case 1: a 56-year-old woman with 20 years of history of pain and swell in elbows, ankles and wrist. Then she was diagnosed as systemic lupus erythematosus (SLE) with glomerulonephritis type III. After other pathologies were ruled out Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and after third infusion a progressive improvement of pain, mobility and daily activities were observed. Case 2: a 70-years-old man with a history of pain and limited motion in the left shoulder without X-ray abnormality. Six months later pathological fracture in the left humerus occurred and after ruled out other pathologies Gorham-Stout disease was diagnosed. Intravenous zoledronic acid (5 mg) was indicated and a good response was observed after the first infusion. Nowadays just over 200 cases were reported. Gorham-Stout disease was reported in different bones, at different age presentation and severe physical deformities, disabilities, and life-threatening complications can occur. Two cases of Gorham-Stout disease with good response to zoledronic acid was reported in this article.

Abstract Image

Abstract Image

唑来膦酸治疗效果良好的Gorham-Stout病2例。
Gorham-Stout病,也称为消失骨综合征,是一种罕见的骨病,其特征是非肿瘤性血管组织的进行性骨内增生导致大量骨溶解。在此,我们报告两例Gorham-Stout病的临床病例。病例1:56岁女性,肘部、脚踝和手腕疼痛和肿胀20年。诊断为系统性红斑狼疮合并III型肾小球肾炎。在排除其他病理后,诊断为Gorham-Stout病。静脉注射唑来膦酸(5mg),第三次输注后,观察到疼痛、活动能力和日常活动的逐渐改善。病例2:70岁男性,左肩疼痛,活动受限,无x线异常。6个月后发生左肱骨病理性骨折,排除其他病理后诊断为Gorham-Stout病。静脉注射唑来膦酸(5mg),首次输注后观察到良好的反应。目前报告的病例只有200多例。据报道,Gorham-Stout病发生在不同的骨骼、不同的年龄、严重的身体畸形、残疾和危及生命的并发症可能发生。本文报道两例对唑来膦酸有良好反应的Gorham-Stout病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
Clinical Cases in Mineral and Bone Metabolism
Clinical Cases in Mineral and Bone Metabolism ENDOCRINOLOGY & METABOLISM-
CiteScore
2.60
自引率
0.00%
发文量
0
期刊介绍: The Journal encourages the submission of case reports and clinical vignettes that provide new and exciting insights into the pathophysiology and characteristics of disorders related to skeletal function and mineral metabolism and/or highlight pratical diagnostic and /or therapeutic considerations.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信