• 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

荷兰的反复出现和创始突变:单一新型反复出现的原纤维蛋白-1 错义突变的马凡综合征患者的广泛临床变异性。

Recurrent and founder mutations in the Netherlands: Extensive clinical variability in Marfan syndrome patients with a single novel recurrent fibrillin-1 missense mutation.

机构信息

Department of Cardiology, Thorax Centre, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.

出版信息

Neth Heart J. 2010 Feb;18(2):85-9. doi: 10.1007/BF03091743.

DOI:10.1007/BF03091743
PMID:20200614
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC2828568/
Abstract

Background/Methods. Marfan syndrome (MFS) is a heritable connective tissue disorder usually caused by a mutation in the fibrillin 1 (FBN1) gene. Typical characteristics of MFS that have been described include dolichostenomelia, ectopia lentis and aortic root dilatation. However, there is great clinical variability in the expression of the syndrome's manifestations, both between and within families. Here we discuss the clinical variability of MFS by describing a large fourgeneration Dutch family with MFS.Results. Nineteen individuals of one family with a single missense FBN1 mutation (c.7916A>G) were identified. The same mutation was found in one unrelated person. Clinical variability was extensive and not all mutation carriers fulfilled the diagnostic criteria for MFS. Some patients only expressed mild skeletal abnormalities, whereas aortic root dilation was present in eight patients, an acute type A aortic dissection was recorded in two other patients, and a mitral valve prolapse was present in eight patients. In some patients cardiac features were not present on initial screening, but did however develop over time.Conclusion. MFS is a clinically highly variable syndrome, which means a meticulous evaluation of suspected cases is crucial. Mutation carriers should be re-evaluated regularly as cardiovascular symptoms may develop over time. (Neth Heart J 2010;18:85-9.).

摘要

背景/方法:马凡综合征(MFS)是一种遗传性结缔组织疾病,通常由原纤维蛋白 1(FBN1)基因的突变引起。MFS 的典型特征包括长骨细长、晶状体异位和主动脉根部扩张。然而,该综合征的临床表现存在很大的临床变异性,无论是在个体之间还是在家族内。在这里,我们通过描述一个具有 MFS 的大型四代荷兰家族来讨论 MFS 的临床变异性。

结果

确定了一个家族中有 19 名个体携带单一错义 FBN1 突变(c.7916A>G)。在一个无关的人中也发现了相同的突变。临床变异性很广泛,并非所有突变携带者都符合 MFS 的诊断标准。一些患者仅表现出轻微的骨骼异常,而 8 名患者存在主动脉根部扩张,2 名其他患者出现急性 A 型主动脉夹层,8 名患者存在二尖瓣脱垂。在一些患者中,心脏特征在初始筛查时不存在,但随着时间的推移会出现。

结论

MFS 是一种临床表现高度可变的综合征,这意味着对疑似病例进行仔细评估至关重要。突变携带者应定期重新评估,因为心血管症状可能会随着时间的推移而发展。(Neth Heart J 2010;18:85-9.)。

相似文献

1
Recurrent and founder mutations in the Netherlands: Extensive clinical variability in Marfan syndrome patients with a single novel recurrent fibrillin-1 missense mutation.荷兰的反复出现和创始突变:单一新型反复出现的原纤维蛋白-1 错义突变的马凡综合征患者的广泛临床变异性。
Neth Heart J. 2010 Feb;18(2):85-9. doi: 10.1007/BF03091743.
2
Cardiovascular characteristics in Marfan syndrome and their relation to the genotype.马凡综合征的心血管特征及其与基因型的关系。
Verh K Acad Geneeskd Belg. 2009;71(6):335-71.
3
Genotype and phenotype analysis of 171 patients referred for molecular study of the fibrillin-1 gene FBN1 because of suspected Marfan syndrome.对171例因疑似马凡综合征而转诊进行原纤维蛋白-1基因FBN1分子研究的患者进行基因型和表型分析。
Arch Intern Med. 2001 Nov 12;161(20):2447-54. doi: 10.1001/archinte.161.20.2447.
4
Missense mutations in FBN1 exons 41 and 42 cause Weill-Marchesani syndrome with thoracic aortic disease and Marfan syndrome.FBN1 外显子 41 和 42 的错义突变导致伴有胸主动脉疾病的马凡综合征和 Weill-Marchesani 综合征。
Am J Med Genet A. 2013 Sep;161A(9):2305-10. doi: 10.1002/ajmg.a.36044. Epub 2013 Jul 29.
5
Mutations of FBN1 and genotype-phenotype correlations in Marfan syndrome and related fibrillinopathies.马凡综合征及相关原纤维蛋白病中FBN1的突变与基因型-表型相关性
Hum Mutat. 2002 Sep;20(3):153-61. doi: 10.1002/humu.10113.
6
The roles of two novel FBN1 gene mutations in the genotype-phenotype correlations of Marfan syndrome and ectopia lentis patients with marfanoid habitus.两个新的FBN1基因突变在马凡综合征和晶状体异位合并马凡体型患者基因型-表型相关性中的作用
Genet Test. 2008 Jun;12(2):325-30. doi: 10.1089/gte.2008.0002.
7
Clinical Characteristics of Marfan Syndrome in Korea.韩国马凡综合征的临床特征
Korean Circ J. 2016 Nov;46(6):841-845. doi: 10.4070/kcj.2016.46.6.841. Epub 2016 Oct 24.
8
The clinical spectrum of complete FBN1 allele deletions.完全 FBN1 等位基因缺失的临床谱。
Eur J Hum Genet. 2011 Mar;19(3):247-52. doi: 10.1038/ejhg.2010.174. Epub 2010 Nov 10.
9
Identification of 29 novel and nine recurrent fibrillin-1 (FBN1) mutations and genotype-phenotype correlations in 76 patients with Marfan syndrome.在76例马凡综合征患者中鉴定出29种新的和9种复发性原纤维蛋白-1(FBN1)突变以及基因型与表型的相关性。
Hum Mutat. 2005 Dec;26(6):529-39. doi: 10.1002/humu.20239.
10
Revised Ghent Criteria is Comparable to Original Diagnostic Criteria for Marfan Syndrome with Increased Ability to Clinically Diagnose Related Disorders.修订后的根特标准与马凡综合征的原始诊断标准相当,临床诊断相关疾病的能力有所提高。
J Med Assoc Thai. 2016 Jan;99(1):34-9.

引用本文的文献

1
Human stem cell models for Marfan syndrome: a .马凡综合征的人类干细胞模型:一种……
Front Cell Dev Biol. 2025 Jan 3;12:1498669. doi: 10.3389/fcell.2024.1498669. eCollection 2024.
2
Next-generation sequencing for diagnosis of thoracic aortic aneurysms and dissections: diagnostic yield, novel mutations and genotype phenotype correlations.用于胸主动脉瘤和夹层诊断的下一代测序:诊断率、新突变及基因型-表型相关性
J Transl Med. 2016 May 4;14(1):115. doi: 10.1186/s12967-016-0870-4.
3
The revised role of TGF-β in aortic aneurysms in Marfan syndrome.转化生长因子-β在马凡综合征主动脉瘤中作用的修订版
Neth Heart J. 2015 Feb;23(2):116-21. doi: 10.1007/s12471-014-0622-0.
4
Multi-scale biomechanical remodeling in aging and genetic mutant murine mitral valve leaflets: insights into Marfan syndrome.衰老和基因突变的小鼠二尖瓣叶中的多尺度生物力学重塑:马凡综合征的见解。
PLoS One. 2012;7(9):e44639. doi: 10.1371/journal.pone.0044639. Epub 2012 Sep 11.
5
Mitral valve disease in Marfan syndrome and related disorders.马凡综合征及相关疾病中的二尖瓣疾病。
J Cardiovasc Transl Res. 2011 Dec;4(6):741-7. doi: 10.1007/s12265-011-9314-y. Epub 2011 Aug 25.

本文引用的文献

1
Prevalence data on all Ghent features in a cross-sectional study of 87 adults with proven Marfan syndrome.一项针对87名经证实患有马凡综合征的成年人的横断面研究中,关于所有根特特征的患病率数据。
Eur J Hum Genet. 2009 Oct;17(10):1222-30. doi: 10.1038/ejhg.2009.30. Epub 2009 Mar 18.
2
Clinical and molecular study of 320 children with Marfan syndrome and related type I fibrillinopathies in a series of 1009 probands with pathogenic FBN1 mutations.在1009例携带致病性FBN1基因突变的先证者系列研究中,对320例患有马凡综合征及相关I型原纤维蛋白病的儿童进行临床和分子研究。
Pediatrics. 2009 Jan;123(1):391-8. doi: 10.1542/peds.2008-0703.
3
Compound-heterozygous Marfan syndrome.复合杂合型马凡综合征
Eur J Med Genet. 2009 Jan-Feb;52(1):1-5. doi: 10.1016/j.ejmg.2008.11.004. Epub 2008 Nov 27.
4
Report of the National Heart, Lung, and Blood Institute and National Marfan Foundation Working Group on research in Marfan syndrome and related disorders.美国国立心肺血液研究所与美国马凡氏综合征基金会关于马凡氏综合征及相关疾病研究的工作组报告。
Circulation. 2008 Aug 12;118(7):785-91. doi: 10.1161/CIRCULATIONAHA.108.783753.
5
Contribution of molecular analyses in diagnosing Marfan syndrome and type I fibrillinopathies: an international study of 1009 probands.分子分析在诊断马凡综合征和I型原纤维蛋白病中的作用:一项对1009名先证者的国际研究
J Med Genet. 2008 Jun;45(6):384-90. doi: 10.1136/jmg.2007.056382. Epub 2008 Feb 29.
6
Utility of molecular analyses in the exploration of extreme intrafamilial variability in the Marfan syndrome.分子分析在探索马凡综合征家族内极端变异性中的应用
Clin Genet. 2007 Sep;72(3):188-98. doi: 10.1111/j.1399-0004.2007.00845.x.
7
Comprehensive genetic analysis of relevant four genes in 49 patients with Marfan syndrome or Marfan-related phenotypes.对49例马凡综合征或马凡相关表型患者的4个相关基因进行全面基因分析。
Am J Med Genet A. 2006 Aug 15;140(16):1719-25. doi: 10.1002/ajmg.a.31353.
8
TGFBR1 and TGFBR2 mutations in patients with features of Marfan syndrome and Loeys-Dietz syndrome.患有马凡氏综合征和洛伊氏综合征特征的患者中的转化生长因子β受体1(TGFBR1)和转化生长因子β受体2(TGFBR2)突变
Hum Mutat. 2006 Aug;27(8):770-7. doi: 10.1002/humu.20354.
9
Losartan, an AT1 antagonist, prevents aortic aneurysm in a mouse model of Marfan syndrome.氯沙坦是一种血管紧张素Ⅱ1型受体(AT1)拮抗剂,可在马方综合征小鼠模型中预防主动脉瘤形成。
Science. 2006 Apr 7;312(5770):117-21. doi: 10.1126/science.1124287.
10
The molecular genetics of Marfan syndrome and related disorders.马凡综合征及相关疾病的分子遗传学
J Med Genet. 2006 Oct;43(10):769-87. doi: 10.1136/jmg.2005.039669. Epub 2006 Mar 29.