Literature DB >> 14613740

Phenotypic variability of cardiovascular manifestations in Marfan Syndrome. Possible role of hyperhomocysteinemia and C677T MTHFR gene polymorphism.

Betti Giusti1, Maria Cristina Porciani, Tamara Brunelli, Lucia Evangelisti, Sandra Fedi, Gian Franco Gensini, Rosanna Abbate, Guido Sani, Magdi Yacoub, Guglielmina Pepe.   

Abstract

AIMS: The aim of this study was to evaluate (1) homocysteinemia and the prevalence of the C677T MTHFR polymorphism in Marfan patients and (2) whether the severity of cardiovascular manifestations is associated with homocysteinemia and/or C677T polymorphism. METHODS AND
RESULTS: We studied 107 patients subdivided into three subgroups based on the severity of cardiovascular manifestations: (A) no involvement (n=4); (B) mild involvement (n=45); (C) aortic dilatation or aortic dissection (n=58), and 189 controls. Total homocysteine (tHcy) was significantly higher in subgroup C than in subgroup B. In subgroup C patients with dissection tHcy was higher than in those without dissection. In subgroup C the prevalence of 677T homozygotes was higher, but not significantly, than in the subgroup B. In patients with dissection the prevalence of 677T homozygotes was significantly higher than in those without dissection and than in subgroup B. In the logistic regression analysis, severe cardiovascular manifestations and aortic dissection in Marfan patients were associated with tHcy plasma levels.
CONCLUSIONS: Our data indicate an association between the severity of the cardiovascular manifestations, in particular aortic dissection, and elevated tHcy levels. This suggests an important role for tHcy in determining phenotypic variability in Marfan patients and provides further evidence for the association of homocysteinemia with the damage of the vascular system.

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Year:  2003        PMID: 14613740     DOI: 10.1016/j.ehj.2003.08.020

Source DB:  PubMed          Journal:  Eur Heart J        ISSN: 0195-668X            Impact factor:   29.983


  20 in total

1.  Homocysteine modifies structural and functional properties of fibronectin and interferes with the fibronectin-fibrillin-1 interaction.

Authors:  Dirk Hubmacher; Laetitia Sabatier; Douglas S Annis; Deane F Mosher; Dieter P Reinhardt
Journal:  Biochemistry       Date:  2011-05-19       Impact factor: 3.162

2.  Moderately Elevated Homocysteine Does Not Contribute to Thoracic Aortic Aneurysm in Mice.

Authors:  Jasmin Roohi; Benjamin Kang; David Bernard; Djahida Bedja; Harry C Dietz; Lawrence C Brody
Journal:  J Nutr       Date:  2017-05-24       Impact factor: 4.798

3.  Recurrent and founder mutations in the Netherlands: Extensive clinical variability in Marfan syndrome patients with a single novel recurrent fibrillin-1 missense mutation.

Authors:  J J J Aalberts; A G Schuurman; G Pals; B J C Hamel; G Bosman; Y Hilhorst-Hofstee; D Q C M Barge-Schaapveld; B J M Mulder; M P van den Berg; J P van Tintelen
Journal:  Neth Heart J       Date:  2010-02       Impact factor: 2.380

4.  Endothelial dysfunction and compromised eNOS/Akt signaling in the thoracic aorta during the progression of Marfan syndrome.

Authors:  A W Y Chung; K Au Yeung; S F Cortes; G G S Sandor; D P Judge; H C Dietz; C van Breemen
Journal:  Br J Pharmacol       Date:  2007-03-05       Impact factor: 8.739

Review 5.  [Imaging of aortic disease].

Authors:  P Reimer; R Vosshenrich; P Landwehr; M Storck
Journal:  Radiologe       Date:  2007-05       Impact factor: 0.635

6.  Functional consequences of homocysteinylation of the elastic fiber proteins fibrillin-1 and tropoelastin.

Authors:  Dirk Hubmacher; Judith T Cirulis; Ming Miao; Fred W Keeley; Dieter P Reinhardt
Journal:  J Biol Chem       Date:  2009-11-04       Impact factor: 5.157

7.  Dysfunction of endothelial and smooth muscle cells in small arteries of a mouse model of Marfan syndrome.

Authors:  H T Syyong; A W Y Chung; H H C Yang; C van Breemen
Journal:  Br J Pharmacol       Date:  2009-10-08       Impact factor: 8.739

Review 8.  Variable severity of cardiovascular phenotypes in patients with an early-onset form of Marfan syndrome harboring FBN1 mutations in exons 24-32.

Authors:  Jun Maeda; Kenjiro Kosaki; Junko Shiono; Kazuki Kouno; Ryo Aeba; Hiroyuki Yamagishi
Journal:  Heart Vessels       Date:  2016-01-21       Impact factor: 2.037

9.  Aortic Dimensions, Biophysical Properties, and Plasma Biomarkers in Children and Adults with Marfan or Loeys-Dietz Syndrome.

Authors:  Jason Z Cui; Kevin C Harris; Koen Raedschelders; Zsuzsanna Hollander; James E Potts; Astrid De Souza; Marla Kiess; Bruce M McManus; Pascal Bernatchez; Leslie A Raffin; Heidi Paine; Cornelis van Breemen; George G S Sandor; Mitra Esfandiarei
Journal:  CJC Open       Date:  2020-12-28

Review 10.  Potential predictors of severe cardiovascular involvement in Marfan syndrome: the emphasized role of genotype-phenotype correlations in improving risk stratification-a literature review.

Authors:  Zoltán Szabolcs; Kálmán Benke; Roland Stengl; Bence Ágg; Miklós Pólos; Gábor Mátyás; Gábor Szabó; Béla Merkely; Tamás Radovits
Journal:  Orphanet J Rare Dis       Date:  2021-05-31       Impact factor: 4.123

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