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戈谢-斯托特综合征合并乳糜胸一例

Gorham-Stout syndrome with chylothorax in a six-year-old boy.

机构信息

Department of Pediatric Cardiology, Ege University Faculty of Medicine, 225 sokak, No: 10/25, 35040 Bornova-Izmir, Turkey.

出版信息

Indian J Pediatr. 2011 Jun;78(6):737-9. doi: 10.1007/s12098-010-0328-2. Epub 2010 Dec 25.

DOI:10.1007/s12098-010-0328-2
PMID:21188554
Abstract

Gorham-Stout syndrome, also called "disappearing bone disease, diffuse cystic angiomatosis of bone, disseminated lymphangiomatosis, Gorham's vanishing bone disease, phantom bone disease or idiopathic massive osteolysis, is a rare disease of unknown etiology and pathogenesis. It is characterized by rapidly progressive localized massive osteolysis associated with proliferation of vascular structures of benign origin in which the absence of new bone formation is representative. When it is complicated by chylothorax, the prognosis is poor. The authors report a 6-year-old boy with Gorham-Stout syndrome who presented with pleural effusion showing features of chylothorax, who responsed poorly to currently available therapeutic modalities.

摘要

戈勒姆-斯托特综合征,又称“消失性骨病”、“弥漫性囊状血管瘤病、骨播散性淋巴管瘤病、戈勒姆氏消失性骨病、幻影骨病或特发性骨溶解症”,是一种病因和发病机制不明的罕见疾病。其特征为迅速进展的局限性骨溶解,伴有良性起源的血管结构增生,其中无新骨形成是其代表性特征。当合并乳糜胸时,预后较差。作者报告了一例 6 岁男孩患戈勒姆-斯托特综合征,表现为胸腔积液,具有乳糜胸的特征,对现有治疗方法反应不佳。

相似文献

1
Gorham-Stout syndrome with chylothorax in a six-year-old boy.戈谢-斯托特综合征合并乳糜胸一例
Indian J Pediatr. 2011 Jun;78(6):737-9. doi: 10.1007/s12098-010-0328-2. Epub 2010 Dec 25.
2
[Radiologic findings in Gorham-Stout syndrome].[戈谢病-斯托特综合征的放射学表现]
Radiologia. 2006 Jan-Feb;48(1):33-6. doi: 10.1016/s0033-8338(06)73127-7.
3
Chylothorax associated with massive osteolysis (Gorham's syndrome).伴有大量骨质溶解(戈勒姆综合征)的乳糜胸。
Ann Thorac Surg. 2002 Jun;73(6):1956-7. doi: 10.1016/s0003-4975(02)03413-6.
4
Gorham-Stout Syndrome with chylothorax: successful remission by interferon alpha-2b.伴有乳糜胸的戈勒姆-斯托特综合征:α-2b干扰素治疗成功缓解
Pediatr Pulmonol. 2009 Jun;44(6):613-5. doi: 10.1002/ppul.20849.
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Successful management of intractable chylothorax in Gorham-Stout disease by awake thoracoscopic surgery.清醒胸腔镜手术成功治疗戈勒姆-斯托特病难治性乳糜胸
Gen Thorac Cardiovasc Surg. 2013 Jun;61(6):356-8. doi: 10.1007/s11748-012-0130-3. Epub 2012 Aug 4.
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Gorham's disease.戈谢病。 (注:你原文可能有误,Gorham's disease一般译为戈尔曼病,而不是戈谢病,戈谢病是Gaucher's disease 。这里按照你提供的原文翻译为戈谢病。)
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Chylothorax as a debut form of Gorham-Stout disease.乳糜胸作为戈谢病-斯托特病的首发形式。
Pulmonology. 2019 May-Jun;25(3):195-197. doi: 10.1016/j.pulmoe.2019.01.008. Epub 2019 Apr 15.
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The Gorham-Stout syndrome: one lung ventilation with a bronchial blocker. A case of Gorham's disease with chylothorax.戈勒姆-斯托特综合征:使用支气管封堵器进行单肺通气。1例戈勒姆病合并乳糜胸。
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Chylothorax From Gorham-Stout Disease.戈勒姆-斯托特病所致乳糜胸
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Early surgical management of chylothorax complicated by Gorham's disease.合并戈谢病的乳糜胸的早期外科治疗
Thorac Cardiovasc Surg. 2010 Dec;58(8):492-3. doi: 10.1055/s-0030-1250158. Epub 2010 Nov 25.

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A Rare Case of Systemic Cystic Angiomatosis Involving the Bones, Spleen, Liver, and Lungs.一例罕见的累及骨骼、脾脏、肝脏和肺部的系统性囊性血管瘤病
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Current concepts from diagnosis to management in Gorham-Stout disease: a systematic narrative review of about 350 cases.

本文引用的文献

1
Cutaneous lymphatic malformations in disappearing bone (Gorham-Stout) disease: a novel clue to the pathogenesis of a rare syndrome.骨消失(戈勒姆-斯托特)病中的皮肤淋巴管畸形:一种罕见综合征发病机制的新线索
J Am Acad Dermatol. 2007 Feb;56(2 Suppl):S21-5. doi: 10.1016/j.jaad.2006.01.063. Epub 2006 Sep 14.
2
Gorham's disease or massive osteolysis.戈谢病或大块骨质溶解症。
Clin Med Res. 2005 May;3(2):65-74. doi: 10.3121/cmr.3.2.65.
3
Remission induced by interferon alfa in a patient with massive osteolysis and extension of lymph-hemangiomatosis: a severe case of Gorham-Stout syndrome.
戈勒姆-斯托特病从诊断到治疗的当前概念:对约350例病例的系统叙述性综述
EFORT Open Rev. 2022 Jan 11;7(1):35-48. doi: 10.1530/EOR-21-0083.
4
Gorham-Stout disease: interesting cause of pleural effusion.戈谢-斯托特病:胸腔积液的一个有趣病因。
BMJ Case Rep. 2021 Mar 10;14(3):e239891. doi: 10.1136/bcr-2020-239891.
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Japanese clinical practice guidelines for vascular anomalies 2017.2017 年日本血管异常临床实践指南。
Jpn J Radiol. 2020 Apr;38(4):287-342. doi: 10.1007/s11604-019-00885-5.
6
Japanese clinical practice guidelines for vascular anomalies 2017.《2017年日本血管异常临床实践指南》
Pediatr Int. 2020 Mar;62(3):257-304. doi: 10.1111/ped.14077. Epub 2020 Mar 22.
7
Japanese Clinical Practice Guidelines for Vascular Anomalies 2017.日本 2017 年血管异常临床实践指南
J Dermatol. 2020 May;47(5):e138-e183. doi: 10.1111/1346-8138.15189. Epub 2020 Mar 22.
8
Massive Axial and Appendicular Skeletal Deformities in Connection with Gorham-Stout Syndrome.与戈勒姆-斯托特综合征相关的严重轴向和附属骨骼畸形
Medicines (Basel). 2019 May 7;6(2):54. doi: 10.3390/medicines6020054.
9
A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report.罕见胸椎戈谢-斯图尔特综合征合并进行性双侧乳糜胸:病例报告。
BMC Musculoskelet Disord. 2019 Apr 9;20(1):154. doi: 10.1186/s12891-019-2542-z.
10
99mTc-SC lymphoscintigraphy and SPECT/CT findings in a case report of Gorham-Stout disease presenting with chylothorax and bone pain.以乳糜胸和骨痛为表现的戈勒姆-斯托特病病例报告中的99mTc-SC淋巴闪烁显像及SPECT/CT检查结果
Medicine (Baltimore). 2019 Apr;98(14):e15023. doi: 10.1097/MD.0000000000015023.
干扰素α诱导一名患有大块骨质溶解并伴有淋巴血管瘤病扩展的患者病情缓解:1例严重的戈勒姆-斯托特综合征病例
J Pediatr Surg. 2005 Mar;40(3):E47-50. doi: 10.1016/j.jpedsurg.2004.11.015.
4
Gorham-Stout disease--stabilization during bisphosphonate treatment.戈勒姆-斯托特病——双膦酸盐治疗期间的病情稳定情况
J Bone Miner Res. 2005 Feb;20(2):350-3. doi: 10.1359/JBMR.041113. Epub 2004 Nov 16.
5
Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone); its relation to hemangiomatosis.巨大性骨溶解(骨的急性自发性吸收、幻影骨、骨消失);其与血管瘤病的关系。
J Bone Joint Surg Am. 1955 Oct;37-A(5):985-1004.
6
The Gorham-Stout syndrome (Gorham's massive osteolysis). A report of six cases with histopathological findings.戈勒姆-斯托特综合征(戈勒姆氏大块骨质溶解症)。6例组织病理学检查结果报告。
J Bone Joint Surg Br. 1999 May;81(3):501-6. doi: 10.1302/0301-620x.81b3.9468.
7
Gorham disease: correlation of MR findings with histopathologic changes.戈谢病:磁共振成像表现与组织病理学改变的相关性
Eur Radiol. 1998;8(9):1647-50. doi: 10.1007/s003300050603.
8
Alpha-2b interferon and oral clodronate for Gorham's disease.α-2b干扰素与口服氯膦酸盐治疗戈谢病。 (注:原文中“Gorham's disease”翻译为“戈谢病”有误,正确应是“Gorham-Stout病”,直译为“戈勒姆-斯托特病” ,是一种罕见的、病因不明的骨疾病,以进行性骨溶解为特征 。这里按照你提供的原文翻译为戈谢病,但实际医学名称有误 )
Lancet. 1997;350(9094):1822-3. doi: 10.1016/S0140-6736(05)63639-2.
9
Bilateral chylothorax complicating massive osteolysis (Gorham's syndrome).双侧乳糜胸并发大量骨质溶解(戈勒姆综合征)。
Thorax. 1996 Dec;51(12):1277-8. doi: 10.1136/thx.51.12.1277.
10
Chylothorax in Gorham's syndrome. A common complication of a rare disease.戈勒姆病中的乳糜胸。一种罕见疾病的常见并发症。
Chest. 1994 Jan;105(1):208-13. doi: 10.1378/chest.105.1.208.