Literature DB >> 16799921

TGFBR1 and TGFBR2 mutations in patients with features of Marfan syndrome and Loeys-Dietz syndrome.

Krishna Kumar Singh1, Kathrin Rommel, Anjali Mishra, Matthias Karck, Axel Haverich, Jörg Schmidtke, Mine Arslan-Kirchner.   

Abstract

Marfan syndrome (MFS) is an autosomal dominant connective tissue disorder characterized by manifestations in the cardiovascular, skeletal, ocular, and other organ systems. MFS type1 (MFS1) is caused by mutations in the gene encoding fibrillin (FBN1). Recently, the transforming growth factor-beta receptor-2 gene, TGFBR2, has been shown to be associated with a second type of this disorder with typically mild or absent ocular involvement (MFS type 2; MFS2). Several point mutations were found in the highly conserved serine/threonine kinase domain of TGFBR2. Mutations in both TGFBR1 and TGFBR2 are associated with Loeys-Dietz aortic aneurysm syndrome (LDS). We searched for TGFBR1 and TGFBR2 mutations in 41 unrelated patients fulfilling the diagnostic criteria of Ghent nosology or with the tentative diagnosis of Marfan syndrome, in whom mutations in the FBN1 coding region were not identified. In TGFBR1, two mutations and two polymorphisms were detected. In TGFBR2, five mutations and six polymorphisms were identified. Reexamination of patients with a TGFBR1 or TGFBR2 mutation revealed extensive clinical overlap between patients with MFS1, MFS2, and LDS.

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Year:  2006        PMID: 16799921     DOI: 10.1002/humu.20354

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  60 in total

1.  Clinical utility gene card for: Marfan syndrome type 1 and related phenotypes [FBN1].

Authors:  Mine Arslan-Kirchner; Eloisa Arbustini; Catherine Boileau; Anne Child; Gwenaelle Collod-Beroud; Anne De Paepe; Jörg Epplen; Guillaume Jondeau; Bart Loeys; Laurence Faivre
Journal:  Eur J Hum Genet       Date:  2010-04-07       Impact factor: 4.246

2.  2006 Curt Stern Award Address. Marfan syndrome: from molecules to medicines.

Authors:  Harry C Dietz
Journal:  Am J Hum Genet       Date:  2007-10       Impact factor: 11.025

3.  Common variants in TGFBR2 and miR-518 genes are associated with hypertension in the Chinese population.

Authors:  Jinfeng Chen; Xianghai Zhao; Hairu Wang; Yanchun Chen; Wen Wang; Wei Zhou; Xuecai Wang; Junming Tang; Yanping Zhao; Xiangfeng Lu; Shufeng Chen; Laiyuan Wang; Chong Shen; Song Yang
Journal:  Am J Hypertens       Date:  2014-03-31       Impact factor: 2.689

Review 4.  Aortic aneurysms: an immune disease with a strong genetic component.

Authors:  Helena Kuivaniemi; Chris D Platsoucas; M David Tilson
Journal:  Circulation       Date:  2008-01-15       Impact factor: 29.690

5.  Report of the National Heart, Lung, and Blood Institute and National Marfan Foundation Working Group on research in Marfan syndrome and related disorders.

Authors:  Gail D Pearson; Richard Devereux; Bart Loeys; Cheryl Maslen; Dianna Milewicz; Reed Pyeritz; Francesco Ramirez; Daniel Rifkin; Lynn Sakai; Lars Svensson; Andy Wessels; Jennifer Van Eyk; Harry C Dietz
Journal:  Circulation       Date:  2008-08-12       Impact factor: 29.690

6.  Sequencing of TGF-beta pathway genes in familial cases of intracranial aneurysm.

Authors:  Teresa Santiago-Sim; Sumy Mathew-Joseph; Hariyadarshi Pannu; Dianna M Milewicz; Christine E Seidman; J G Seidman; Dong H Kim
Journal:  Stroke       Date:  2009-03-19       Impact factor: 7.914

7.  Tgfbr2 is required in osterix expressing cells for postnatal skeletal development.

Authors:  Sarah B Peters; Ying Wang; Rosa Serra
Journal:  Bone       Date:  2016-12-30       Impact factor: 4.398

8.  Clinical utility gene card for: Loeys-Dietz syndrome (TGFBR1/2) and related phenotypes.

Authors:  Mine Arslan-Kirchner; Jörg T Epplen; Laurence Faivre; Guillaume Jondeau; Jörg Schmidtke; Anne De Paepe; Bart Loeys
Journal:  Eur J Hum Genet       Date:  2011-04-27       Impact factor: 4.246

9.  TGFβRIIb mutations trigger aortic aneurysm pathogenesis by altering transforming growth factor β2 signal transduction.

Authors:  Katharine J Bee; David C Wilkes; Richard B Devereux; Craig T Basson; Cathy J Hatcher
Journal:  Circ Cardiovasc Genet       Date:  2012-10-24

10.  Prevalence data on all Ghent features in a cross-sectional study of 87 adults with proven Marfan syndrome.

Authors:  Svend Rand-Hendriksen; Rigmor Lundby; Lena Tjeldhorn; Kai Andersen; Jon Offstad; Svein Ove Semb; Hans-Jørgen Smith; Benedicte Paus; Odd Geiran
Journal:  Eur J Hum Genet       Date:  2009-03-18       Impact factor: 4.246

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