• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

肥厚型心肌病发现的故事讲述

Storytelling of Hypertrophic Cardiomyopathy Discovery.

作者信息

Thiene Gaetano, Calore Chiara, De Gaspari Monica, Basso Cristina

机构信息

Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua Medical School, 35121 Padova, Italy.

出版信息

J Cardiovasc Dev Dis. 2024 Sep 28;11(10):300. doi: 10.3390/jcdd11100300.

DOI:10.3390/jcdd11100300
PMID:39452271
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11508572/
Abstract

The discovery of hypertrophic cardiomyopathy (HCM) dates back to 1958, when the pathologist Donald Teare of the St. George's Hospital in London performed autopsies in eight cases with asymmetric hypertrophy of the ventricular septum and bizarre disorganization (disarray) at histology, first interpreted as hamartoma. Seven had died suddenly. The cardiac specimens were cut along the long axis, similar to the 2D echo. In the same year, at the National Institute of Health U.S.A., Eugene Braunwald, a hemodynamist, and Andrew Glenn Morrow, a cardiac surgeon, clinically faced a patient with an apparently similar morbid entity, with a systolic murmur and subaortic valve gradient. "Discrete" subaortic stenosis was postulated. However, at surgery, Dr. Morrow observed only hypertrophy and performed myectomy to relieve the obstruction. This first Braunwald-Morrow patient underwent a successful cardiac transplant later at the disease end stage. The same Dr. Morrow was found to be affected by the familial HCM and died suddenly in 1992. The term "functional subaortic stenosis" was used in 1959 and "idiopathic hypertrophic subaortic stenosis" in 1960. Years before, in 1957, Lord Brock, a cardiac surgeon at the Guy's Hospital in London, during alleged aortic valve surgery in extracorporeal circulation, did not find any valvular or discrete subaortic stenoses. In 1980, John F. Goodwin of the Westminster Hospital in London, the head of an international WHO committee, put forward the first classification of heart muscle diseases, introducing the term cardiomyopathy (dilated, hypertrophic, and endomyocardial restrictive). In 1995, the WHO classification was revisited, with the addition of two new entities, namely arrhythmogenic and purely myocardial restrictive, the latter a paradox of a small heart accounting for severe congestive heart failure by ventricular diastolic impairment. A familial occurrence was noticed earlier in HCM and published by Teare and Goodwin in 1960. In 1989-1990, the same family underwent molecular genetics investigation by the Seidman team in Boston, and a missense mutation of the β-cardiac myosin heavy chain in chromosome 14 was found. Thus, 21 years elapsed from HCM gross discovery to molecular discoveries. The same original family was the source of both the gross and genetic explanations of HCM, which is now named sarcomere disease. Restrictive cardiomyopathy, characterized grossly without hypertrophy and histologically by myocardial disarray, was found to also have a sarcomeric genetic mutation, labeled "HCM without hypertrophy". Sarcomere missense mutations have also been reported in dilated cardiomyopathy (DCM) and non-compaction cardiomyopathy. Moreover, sarcomeric gene defects have been detected in some DNA non-coding regions of HCM patients. The same mutation in the family may express different phenotypes (HCM, DCM, and RCM). Large ischemic scars have been reported by pathologists and are nowadays easily detectable in vivo by cardiac magnetic resonance with gadolinium. The ischemic arrhythmic substrate enhances the risk of sudden death.

摘要

肥厚型心肌病(HCM)的发现可追溯到1958年,当时伦敦圣乔治医院的病理学家唐纳德·蒂尔对8例室间隔不对称肥厚且组织学上有奇异紊乱(排列紊乱)的病例进行了尸检,最初被解释为错构瘤。其中7例猝死。心脏标本沿长轴切开,类似于二维超声心动图。同年,在美国国立卫生研究院,血液动力学家尤金·布劳恩瓦尔德和心脏外科医生安德鲁·格伦·莫罗临床上遇到了一名患有明显类似病态实体的患者,有收缩期杂音和主动脉瓣下梯度。推测为“离散性”主动脉瓣下狭窄。然而,在手术中,莫罗医生仅观察到肥厚,并进行了心肌切除术以解除梗阻。这位首位布劳恩瓦尔德 - 莫罗患者在疾病终末期后来接受了成功的心移植手术。发现同一位莫罗医生患有家族性HCM,并于1992年猝死。1959年使用了“功能性主动脉瓣下狭窄”一词,1960年使用了“特发性肥厚性主动脉瓣下狭窄”一词。早在1957年,伦敦盖伊医院的心脏外科医生布罗克勋爵在所谓的体外循环主动脉瓣手术中,未发现任何瓣膜或离散性主动脉瓣下狭窄。1980年,伦敦威斯敏斯特医院的约翰·F·古德温,作为世界卫生组织一个国际委员会的负责人,提出了心肌疾病的首个分类,引入了心肌病(扩张型、肥厚型和心内膜限制性)这一术语。1995年,对世界卫生组织的分类进行了修订,增加了两个新的实体,即致心律失常性和单纯心肌限制性,后者是一种心脏小却因心室舒张功能障碍导致严重充血性心力衰竭的矛盾情况。HCM中更早注意到有家族性发病情况,并由蒂尔和古德温于1960年发表。1989 - 1990年,同一个家族在波士顿由塞德曼团队进行了分子遗传学研究,发现了14号染色体上β - 心脏肌球蛋白重链的一个错义突变。因此,从HCM的大体发现到分子发现历经了21年。同一个原始家族是HCM大体和遗传学解释的来源,HCM现在被称为肌节病。限制性心肌病,大体上无肥厚特征,组织学上有心肌排列紊乱,也被发现有肌节基因突变,标记为“无肥厚的HCM”。在扩张型心肌病(DCM)和非致密化心肌病中也报道了肌节错义突变。此外,在HCM患者的一些DNA非编码区域检测到了肌节基因缺陷。家族中的相同突变可能表现出不同的表型(HCM、DCM和RCM)。病理学家报告了大的缺血性瘢痕,如今通过钆增强心脏磁共振成像在体内很容易检测到。缺血性心律失常基质增加了猝死风险。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/7c91a2470f64/jcdd-11-00300-g022.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/50f9d8baca70/jcdd-11-00300-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6badd38dba1e/jcdd-11-00300-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/8f2016827efc/jcdd-11-00300-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/723bd894b2b0/jcdd-11-00300-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/84659d7398e4/jcdd-11-00300-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/77b730f32ac1/jcdd-11-00300-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/796f54ab0f2f/jcdd-11-00300-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/7e9f643e376b/jcdd-11-00300-g008.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/e6ccebd2873b/jcdd-11-00300-g009.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/39352766ed08/jcdd-11-00300-g010.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/02b31e14aeaf/jcdd-11-00300-g011.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/425b56d827ec/jcdd-11-00300-g012.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6b5b837cc571/jcdd-11-00300-g013.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/14ef20195915/jcdd-11-00300-g014.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/202b28583278/jcdd-11-00300-g015.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/8a8545b357c8/jcdd-11-00300-g016.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/b26646e68828/jcdd-11-00300-g017.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/044c26dedeff/jcdd-11-00300-g018.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/da732a83c1dd/jcdd-11-00300-g019.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/ec00df8dba50/jcdd-11-00300-g020.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6e664643f0ba/jcdd-11-00300-g021.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/7c91a2470f64/jcdd-11-00300-g022.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/50f9d8baca70/jcdd-11-00300-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6badd38dba1e/jcdd-11-00300-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/8f2016827efc/jcdd-11-00300-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/723bd894b2b0/jcdd-11-00300-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/84659d7398e4/jcdd-11-00300-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/77b730f32ac1/jcdd-11-00300-g006.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/796f54ab0f2f/jcdd-11-00300-g007.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/7e9f643e376b/jcdd-11-00300-g008.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/e6ccebd2873b/jcdd-11-00300-g009.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/39352766ed08/jcdd-11-00300-g010.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/02b31e14aeaf/jcdd-11-00300-g011.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/425b56d827ec/jcdd-11-00300-g012.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6b5b837cc571/jcdd-11-00300-g013.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/14ef20195915/jcdd-11-00300-g014.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/202b28583278/jcdd-11-00300-g015.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/8a8545b357c8/jcdd-11-00300-g016.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/b26646e68828/jcdd-11-00300-g017.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/044c26dedeff/jcdd-11-00300-g018.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/da732a83c1dd/jcdd-11-00300-g019.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/ec00df8dba50/jcdd-11-00300-g020.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/6e664643f0ba/jcdd-11-00300-g021.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9933/11508572/7c91a2470f64/jcdd-11-00300-g022.jpg

相似文献

1
Storytelling of Hypertrophic Cardiomyopathy Discovery.肥厚型心肌病发现的故事讲述
J Cardiovasc Dev Dis. 2024 Sep 28;11(10):300. doi: 10.3390/jcdd11100300.
2
Surgical pathology of subaortic septal myectomy: histology skips over clinical diagnosis.主动脉瓣下间隔心肌切除术的外科病理学:组织学跳过临床诊断。
Cardiovasc Pathol. 2018 Mar-Apr;33:32-38. doi: 10.1016/j.carpath.2017.12.002. Epub 2018 Jan 3.
3
[Classification of cardiomyopathies and indication for endomyocardial biopsy revisited].[心肌病的分类及心内膜心肌活检指征再探讨]
Herz. 2009 Feb;34(1):55-62. doi: 10.1007/s00059-009-3195-8.
4
Hypertrophic Cardiomyopathy: Genetics, Pathogenesis, Clinical Manifestations, Diagnosis, and Therapy.肥厚型心肌病:遗传学、发病机制、临床表现、诊断与治疗
Circ Res. 2017 Sep 15;121(7):749-770. doi: 10.1161/CIRCRESAHA.117.311059.
5
Surgical pathology of subaortic septal myectomy not associated with hypertrophic cardiomyopathy: a study of 98 cases (1996-2000).与肥厚型心肌病无关的主动脉下隔膜切除术的外科病理学:98例研究(1996 - 2000年)
Cardiovasc Pathol. 2003 Jul-Aug;12(4):207-15. doi: 10.1016/s1054-8807(03)00057-7.
6
Cardiac troponin T mutation in familial cardiomyopathy with variable remodeling and restrictive physiology.家族性心肌病伴可变重塑和限制性生理学中的心肌肌钙蛋白T突变
Clin Genet. 2008 Nov;74(5):445-54. doi: 10.1111/j.1399-0004.2008.01062.x. Epub 2008 Jul 21.
7
An Update on Pediatric Cardiomyopathy.小儿心肌病的最新进展
Curr Treat Options Cardiovasc Med. 2019 Jun 25;21(8):36. doi: 10.1007/s11936-019-0739-y.
8
Muscle LIM Protein Force-Sensing Mediates Sarcomeric Biomechanical Signaling in Human Familial Hypertrophic Cardiomyopathy.肌 LIM 蛋白力感介导人类家族性肥厚型心肌病中的肌节生物力学信号转导。
Circulation. 2022 Apr 19;145(16):1238-1253. doi: 10.1161/CIRCULATIONAHA.121.056265. Epub 2022 Apr 6.
9
β-Myosin heavy chain variant Val606Met causes very mild hypertrophic cardiomyopathy in mice, but exacerbates HCM phenotypes in mice carrying other HCM mutations.β-肌球蛋白重链变异 Val606Met 导致小鼠出现非常轻微的肥厚型心肌病,但会加重携带其他 HCM 突变的小鼠的 HCM 表型。
Circ Res. 2014 Jul 7;115(2):227-37. doi: 10.1161/CIRCRESAHA.115.303178. Epub 2014 May 14.
10
[Hypertrophic cardiomyopathy with left ventricular dilatation].[伴有左心室扩张的肥厚型心肌病]
J Cardiol. 1988 Jun;18(2):319-28.

引用本文的文献

1
Hypertrophic Cardiomyopathy: New Clinical and Therapeutic Perspectives of an "Old" Genetic Myocardial Disease.肥厚型心肌病:一种“古老”的遗传性心肌病的新临床与治疗视角
Genes (Basel). 2025 Jan 10;16(1):74. doi: 10.3390/genes16010074.

本文引用的文献

1
The promise of gene therapy in hypertrophic cardiomyopathy: emergence of human experience.基因治疗肥厚型心肌病的前景:人类经验的浮现
Cardiovasc Res. 2024 Jul 2;120(8):e33-e35. doi: 10.1093/cvr/cvae107.
2
Aficamten for Symptomatic Obstructive Hypertrophic Cardiomyopathy.肥厚型梗阻性心肌病的治疗药物:Aficamten
N Engl J Med. 2024 May 30;390(20):1849-1861. doi: 10.1056/NEJMoa2401424. Epub 2024 May 13.
3
Mechanisms of ischaemia-induced arrhythmias in hypertrophic cardiomyopathy: a large-scale computational study.肥厚型心肌病缺血性心律失常的机制:大规模计算研究。
Cardiovasc Res. 2024 Jul 2;120(8):914-926. doi: 10.1093/cvr/cvae086.
4
Characterization of heterozygous and homozygous mouse models with the most common hypertrophic cardiomyopathy mutation MYBPC3 in the Netherlands.荷兰最常见的肥厚型心肌病突变 MYBPC3 的杂合子和纯合子小鼠模型的特征。
J Mol Cell Cardiol. 2023 Dec;185:65-76. doi: 10.1016/j.yjmcc.2023.10.008. Epub 2023 Oct 14.
5
A cost-effectiveness analysis of hypertrophic cardiomyopathy sudden cardiac death risk algorithms for implantable cardioverter defibrillator decision-making.肥厚型心肌病心脏性猝死风险算法在植入式心脏复律除颤器决策中的成本效益分析。
Eur Heart J Qual Care Clin Outcomes. 2024 Jun 20;10(4):285-293. doi: 10.1093/ehjqcco/qcad050.
6
Treatment Strategies for Cardiomyopathy in Children: A Scientific Statement From the American Heart Association.儿童心肌病的治疗策略:美国心脏协会的科学声明
Circulation. 2023 Jul 11;148(2):174-195. doi: 10.1161/CIR.0000000000001151. Epub 2023 Jun 8.
7
The role of noncoding genetic variants in cardiomyopathy.非编码基因变异在心肌病中的作用。
Front Cardiovasc Med. 2023 May 22;10:1116925. doi: 10.3389/fcvm.2023.1116925. eCollection 2023.
8
Reflections on Hypertrophic Cardiomyopathy.肥厚型心肌病之思考
Eur Heart J. 2021 Aug 17;42(31):2969-2970. doi: 10.1093/eurheartj/ehab337.
9
Mavacamten for treatment of symptomatic obstructive hypertrophic cardiomyopathy (EXPLORER-HCM): a randomised, double-blind, placebo-controlled, phase 3 trial.马卡丹特治疗有症状梗阻性肥厚型心肌病(EXPLORER-HCM)的随机、双盲、安慰剂对照、3 期临床试验
Lancet. 2020 Sep 12;396(10253):759-769. doi: 10.1016/S0140-6736(20)31792-X. Epub 2020 Aug 29.
10
Hypertrophic Cardiomyopathy: Genetics, Pathogenesis, Clinical Manifestations, Diagnosis, and Therapy.肥厚型心肌病:遗传学、发病机制、临床表现、诊断与治疗
Circ Res. 2017 Sep 15;121(7):749-770. doi: 10.1161/CIRCRESAHA.117.311059.