Literature DB >> 17936924

May TGFBR1 act also as low penetrance allele in Marfan syndrome?

Laura Lucarini, Lucia Evangelisti, Monica Attanasio, Ilaria Lapini, Francesca Chiarini, Maria Cristina Porciani, Rosanna Abbate, GianFranco Gensini, Guglielmina Pepe.   

Abstract

Marfan syndrome, a human disease involving cardiovascular and skeletal apparatuses and ocular and central nervous systems, is associated to mutations in FBN1 gene; heterozygous mutations in TGFBR2 and TGFBR1 genes were found associated to MFS type 2, characterized by the presence of skeletal and cardiovascular major criteria and absence of eye major criterion. We screened the TGFBR1 gene in 46 Marfan patients in whom mutations in FBN1 and TGFBR2 genes were excluded and the analysis of Ex1 was extended to additional 114 Marfan patients and 237 controls. We detected two potentially pathological sequence variants: the TGFBR1 6Ala allele whose frequency was higher in the group of Marfan patients (0.13) than in the controls (0.08) (p=0.013; OR=1.69) and an insertion of 20 nucleotides in the 5'UTR that turned out to be a familial silent rare polymorphism. We hypothesize that TGFBR1 sequence variants may act not only as major, but also as low penetrance alleles of the clinical phenotype in Marfan syndrome.

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Year:  2007        PMID: 17936924     DOI: 10.1016/j.ijcard.2007.07.048

Source DB:  PubMed          Journal:  Int J Cardiol        ISSN: 0167-5273            Impact factor:   4.164


  6 in total

Review 1.  Preventing the aortic complications of Marfan syndrome: a case-example of translational genomic medicine.

Authors:  Alain Li-Wan-Po; Bart Loeys; Peter Farndon; David Latham; Caroline Bradley
Journal:  Br J Clin Pharmacol       Date:  2011-07       Impact factor: 4.335

2.  High prevalence of eosinophilic esophagitis in patients with inherited connective tissue disorders.

Authors:  J Pablo Abonia; Ting Wen; Emily M Stucke; Tommie Grotjan; Molly S Griffith; Katherine A Kemme; Margaret H Collins; Philip E Putnam; James P Franciosi; Karl F von Tiehl; Brad T Tinkle; Keith A Marsolo; Lisa J Martin; Stephanie M Ware; Marc E Rothenberg
Journal:  J Allergy Clin Immunol       Date:  2013-04-19       Impact factor: 10.793

3.  The genetics and genomics of thoracic aortic disease.

Authors:  Pawel Pomianowski; John A Elefteriades
Journal:  Ann Cardiothorac Surg       Date:  2013-05

Review 4.  Marfan syndrome: current perspectives.

Authors:  Guglielmina Pepe; Betti Giusti; Elena Sticchi; Rosanna Abbate; Gian Franco Gensini; Stefano Nistri
Journal:  Appl Clin Genet       Date:  2016-05-09

5.  New population-based exome data question the pathogenicity of some genetic variants previously associated with Marfan syndrome.

Authors:  Ren-Qiang Yang; Javad Jabbari; Xiao-Shu Cheng; Reza Jabbari; Jonas B Nielsen; Bjarke Risgaard; Xu Chen; Ahmad Sajadieh; Stig Haunsø; Jesper Hastrup Svendsen; Morten S Olesen; Jacob Tfelt-Hansen
Journal:  BMC Genet       Date:  2014-06-18       Impact factor: 2.797

Review 6.  The Molecular Genetics of Marfan Syndrome.

Authors:  Qiu Du; Dingding Zhang; Yue Zhuang; Qiongrong Xia; Taishen Wen; Haiping Jia
Journal:  Int J Med Sci       Date:  2021-05-27       Impact factor: 3.738

  6 in total

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