Literature DB >> 25337071

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

E Martínez-Quintana1, F Rodríguez-González2, P Garay-Sánchez3, A Tugores3.   

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.

Entities:  

Keywords:  Aorta; Ectopia lentis; Fibrillin 1; Marfan

Year:  2014        PMID: 25337071      PMCID: PMC4188161          DOI: 10.1159/000358846

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  17 in total

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Journal:  Am J Med Genet A       Date:  2004-04-30       Impact factor: 2.802

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  2 in total

1.  Novel Marfan Syndrome-Associated Mutation in the FBN1 Gene Caused by Parental Mosaicism and Leading to Abnormal Limb Patterning.

Authors:  Efrén Martínez-Quintana; Noemí Caballero-Sánchez; Fayna Rodríguez-González; Paloma Garay-Sánchez; Antonio Tugores
Journal:  Mol Syndromol       Date:  2017-03-31

2.  Steered molecular dynamic simulations reveal Marfan syndrome mutations disrupt fibrillin-1 cbEGF domain mechanosensitive calcium binding.

Authors:  Stephen J Haller; Adrian E Roitberg; Andrew T Dudley
Journal:  Sci Rep       Date:  2020-10-08       Impact factor: 4.996

  2 in total

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