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一种导致具有轻微心脏特征的马凡综合征的新型原纤蛋白1基因突变。

A novel fibrillin 1 gene mutation leading to marfan syndrome with minimal cardiac features.

作者信息

Martínez-Quintana E, Rodríguez-González F, Garay-Sánchez P, Tugores A

机构信息

Cardiology Service, Complejo Hospitalario Universitario Insular-Materno Infantil, Las Palmas de Gran Canaria, Spain.

Ophthamology Service, Hospital Universitario Dr. Negrín, Las Palmas de Gran Canaria, Spain.

出版信息

Mol Syndromol. 2014 Aug;5(5):236-40. doi: 10.1159/000358846. Epub 2014 Feb 27.

Abstract

Marfan syndrome is an autosomal dominant disorder of the connective tissue, characterized by early development of thoracic aortic aneurysms and/or dissections, accompanied by ocular and/or skeletal involvement, and is caused by mutations in the fibrillin 1 (FBN1) gene. We report on a patient with ectopia lentis and a nonprogressive aortic root dilatation who presented with a novel mutation affecting a conserved cysteine residue present in a calcium-binding epidermal growth factor-like domain of FBN1 (ENSP00000325527, p.Cys538Phe; Chr15:48,805,751 G>T), as revealed by complete sequencing of the FBN1 gene exons and flanking sequences. Identification of the mutation led to genetic screening of apparently asymptomatic family members, allowing the detection of characteristic ocular phenotypes in the absence of typical cardiac Marfan features. This finding stresses the importance of genetic screening of asymptomatic relatives for FBN1 gene mutation carriers.

摘要

马凡综合征是一种常染色体显性结缔组织疾病,其特征为胸主动脉瘤和/或夹层的早期发展,并伴有眼部和/或骨骼受累,由原纤蛋白1(FBN1)基因突变引起。我们报告了一名患有晶状体异位和非进行性主动脉根部扩张的患者,通过对FBN1基因外显子和侧翼序列进行完整测序发现,该患者存在一种新的突变,影响FBN1钙结合表皮生长因子样结构域中一个保守的半胱氨酸残基(ENSP00000325527,p.Cys538Phe;Chr15:48,805,751 G>T)。该突变的鉴定促使对明显无症状的家庭成员进行基因筛查,从而在缺乏典型心脏马凡特征的情况下检测到特征性眼部表型。这一发现强调了对FBN1基因突变携带者的无症状亲属进行基因筛查的重要性。

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The new Ghent criteria for Marfan syndrome: what do they change?新的马凡综合征根特标准:它们改变了什么?
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The revised Ghent nosology for the Marfan syndrome.修订版马凡综合征根特分类法。
J Med Genet. 2010 Jul;47(7):476-85. doi: 10.1136/jmg.2009.072785.
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Current concepts of ocular manifestations in Marfan syndrome.马凡综合征眼部表现的当前概念。
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Marfan's syndrome.马方综合征
Lancet. 2005 Dec 3;366(9501):1965-76. doi: 10.1016/S0140-6736(05)67789-6.
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Ectopia lentis phenotypes and the FBN1 gene.晶状体异位表型与FBN1基因
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