Literature DB >> 27724990

Identification of FBN1 gene mutations in Ukrainian Marfan syndrome patients.

Rustam Zhurayev1, Dorien Proost2, Dmytro Zerbino1, Viktor Fedorenko3, Josephina A N Meester2, Lut VAN Laer2, Bart L Loeys2.   

Abstract

Marfan syndrome is an autosomal dominant connective tissue disorder, predominantly affecting the ocular, skeletal and cardiovascular systems. Here, we present the results of the first genetic testing in 40 Ukrainian Marfan (-like) patients and 10 relatives. We applied a targeted next generation sequencing panel comprising FBN1 and 13 thoracic aortic aneurysm genes. We identified 27 causal mutations in FBN1, obtaining a mutation yield of 67·5%. A significant difference in age at aortic surgery between mutation positive and negative patients was observed. Thus, we conclude that genetic testing is important to identify patients at higher risk for developing life-threatening cardiovascular complications.

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Year:  2016        PMID: 27724990      PMCID: PMC6865158          DOI: 10.1017/S0016672316000112

Source DB:  PubMed          Journal:  Genet Res (Camb)        ISSN: 0016-6723            Impact factor:   1.588


  19 in total

1.  A comparison of the Ghent and revised Ghent nosologies for the diagnosis of Marfan syndrome in an adult Korean population.

Authors:  Jeong Hoon Yang; Hyejin Han; Shin Yi Jang; Ju Ryoung Moon; Kiick Sung; Tae-Young Chung; Heung Jae Lee; Chang-Seok Ki; Duk-Kyung Kim
Journal:  Am J Med Genet A       Date:  2011-12-07       Impact factor: 2.802

2.  A marked decrease in heart rate variability in Marfan syndrome patients with confirmed FBN1 mutations.

Authors:  Andriy Cherkas; Rustam Zhuraev
Journal:  Cardiol J       Date:  2015-10-27       Impact factor: 2.737

3.  Analysis of phenotype and genotype information for the diagnosis of Marfan syndrome.

Authors:  S Sheikhzadeh; C Kade; B Keyser; M Stuhrmann; M Arslan-Kirchner; M Rybczynski; A M Bernhardt; C R Habermann; M Hillebrand; T Mir; P N Robinson; J Berger; C Detter; S Blankenberg; J Schmidtke; Y von Kodolitsch
Journal:  Clin Genet       Date:  2011-10-05       Impact factor: 4.438

4.  Revised diagnostic criteria for the Marfan syndrome.

Authors:  A De Paepe; R B Devereux; H C Dietz; R C Hennekam; R E Pyeritz
Journal:  Am J Med Genet       Date:  1996-04-24

5.  The new Ghent criteria for Marfan syndrome: what do they change?

Authors:  L Faivre; G Collod-Beroud; L Adès; E Arbustini; A Child; B L Callewaert; B Loeys; C Binquet; E Gautier; K Mayer; M Arslan-Kirchner; M Grasso; C Beroud; D Hamroun; C Bonithon-Kopp; H Plauchu; P N Robinson; J De Backer; P Coucke; U Francke; O Bouchot; J E Wolf; C Stheneur; N Hanna; D Detaint; A De Paepe; C Boileau; G Jondeau
Journal:  Clin Genet       Date:  2011-06-02       Impact factor: 4.438

6.  The revised Ghent nosology for the Marfan syndrome.

Authors:  Bart L Loeys; Harry C Dietz; Alan C Braverman; Bert L Callewaert; Julie De Backer; Richard B Devereux; Yvonne Hilhorst-Hofstee; Guillaume Jondeau; Laurence Faivre; Dianna M Milewicz; Reed E Pyeritz; Paul D Sponseller; Paul Wordsworth; Anne M De Paepe
Journal:  J Med Genet       Date:  2010-07       Impact factor: 6.318

7.  Diagnostic yield in adults screened at the Marfan outpatient clinic using the 1996 and 2010 Ghent nosologies.

Authors:  Jan J J Aalberts; Chris H L Thio; Agnes G Schuurman; Irene M van Langen; Bert A E van der Pol; J Peter van Tintelen; Maarten P van den Berg
Journal:  Am J Med Genet A       Date:  2012-03-27       Impact factor: 2.802

8.  Dysregulation of TGF-beta activation contributes to pathogenesis in Marfan syndrome.

Authors:  Enid R Neptune; Pamela A Frischmeyer; Dan E Arking; Loretha Myers; Tracie E Bunton; Barbara Gayraud; Francesco Ramirez; Lynn Y Sakai; Harry C Dietz
Journal:  Nat Genet       Date:  2003-02-24       Impact factor: 38.330

9.  [Marfan syndrome: clinical and pathomorphological restructurings after surgical treatment of aortic aneurysm].

Authors:  R K Zhuraev; D D Zerbino
Journal:  Angiol Sosud Khir       Date:  2014

10.  dbNSFP: a lightweight database of human nonsynonymous SNPs and their functional predictions.

Authors:  Xiaoming Liu; Xueqiu Jian; Eric Boerwinkle
Journal:  Hum Mutat       Date:  2011-08       Impact factor: 4.878

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