Literature DB >> 18368697

SCN5A variants in Japanese patients with left ventricular noncompaction and arrhythmia.

Lishen Shan1, Naomasa Makita, Yanlin Xing, Sayake Watanabe, Takeshi Futatani, Fei Ye, Kazuyoshi Saito, Keijiro Ibuki, Kazuhiro Watanabe, Keiichi Hirono, Keiichiro Uese, Fukiko Ichida, Toshio Miyawaki, Hideki Origasa, Neil E Bowles, Jeffrey A Towbin.   

Abstract

Left ventricular noncompaction (LVNC) is a genetically heterogenous disorder. Mutations in the human cardiac sodium channel alpha-subunit gene (SCN5A) are involved in the pathophysiology of cardiac arrhythmias and cardiomyopathies. This study was performed to compare the frequency of SCN5A variants in LVNC patients with or without arrhythmias, and to investigate the relationship between variants and disease severity. DNA was isolated from the peripheral blood of 62 Japanese probands with LVNC, comprising 17 familial cases and 45 sporadic cases. Blood samples were screened for variants in SCN5A using single-strand conformational polymorphism analysis (SSCP) and DNA sequencing. Seven variants, rs6599230:G > A, c.453C > T, c.1141-3C > A, rs1805124:A > G (p.H558R), rs1805125:C > T (p.P1090L), c.3996C > T, and rs1805126:T > C were identified in 7 familial and 12 sporadic cases. The frequency of SCN5A variants was significantly higher in the patients with arrhythmias than those without (50% vs 7%: P = 0.0003), suggesting these variants represent a risk factor for arrhythmia and supporting the hypothesis that genes encoding ion channels are involved in LVNC pathophysiology. The LVNC patients with heart failure also had high occurrence of SCN5A variants, suggesting the presence of SCN5A variants and/or arrhythmias increase the severity of LVNC.

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Year:  2008        PMID: 18368697     DOI: 10.1016/j.ymgme.2007.10.009

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  23 in total

Review 1.  Left ventricular noncompaction cardiomyopathy: cardiac, neuromuscular, and genetic factors.

Authors:  Josef Finsterer; Claudia Stöllberger; Jeffrey A Towbin
Journal:  Nat Rev Cardiol       Date:  2017-01-12       Impact factor: 32.419

Review 2.  Left Ventricular Non-compaction: Is It Genetic?

Authors:  Teck Wah Ting; Saumya Shekhar Jamuar; Maggie Siewyan Brett; Ee Shien Tan; Breana Wen Min Cham; Jiin Ying Lim; Hai Yang Law; Ene Choo Tan; Jonathan Tze Liang Choo; Angeline Hwei Meeng Lai
Journal:  Pediatr Cardiol       Date:  2015-06-25       Impact factor: 1.655

Review 3.  Genetics and disease of ventricular muscle.

Authors:  Diane Fatkin; Christine E Seidman; Jonathan G Seidman
Journal:  Cold Spring Harb Perspect Med       Date:  2014-01-01       Impact factor: 6.915

Review 4.  Translating emerging molecular genetic insights into clinical practice in inherited cardiomyopathies.

Authors:  Babken Asatryan; Argelia Medeiros-Domingo
Journal:  J Mol Med (Berl)       Date:  2018-08-20       Impact factor: 4.599

5.  Novel Genetic Triggers and Genotype-Phenotype Correlations in Patients With Left Ventricular Noncompaction.

Authors:  Karol Miszalski-Jamka; John L Jefferies; Wojciech Mazur; Jan Głowacki; Jianhong Hu; Monika Lazar; Richard A Gibbs; Jacek Liczko; Jan Kłyś; Eric Venner; Donna M Muzny; Jarosław Rycaj; Jacek Białkowski; Ewa Kluczewska; Zbigniew Kalarus; Shalini Jhangiani; Hussein Al-Khalidi; Tomasz Kukulski; James R Lupski; William J Craigen; Matthew N Bainbridge
Journal:  Circ Cardiovasc Genet       Date:  2017-08

6.  Intrafamilial variability for novel TAZ gene mutation: Barth syndrome with dilated cardiomyopathy and heart failure in an infant and left ventricular noncompaction in his great-uncle.

Authors:  Diti Ronvelia; Jaclyn Greenwood; Julia Platt; Simin Hakim; Michael V Zaragoza
Journal:  Mol Genet Metab       Date:  2012-09-18       Impact factor: 4.797

Review 7.  Cardiogenetics, neurogenetics, and pathogenetics of left ventricular hypertrabeculation/noncompaction.

Authors:  Josef Finsterer
Journal:  Pediatr Cardiol       Date:  2009-01-29       Impact factor: 1.655

8.  The genetics of cardiomyopathy: genotyping and genetic counseling.

Authors:  Steven J Fowler; Carlo Napolitano; Silvia G Priori
Journal:  Curr Treat Options Cardiovasc Med       Date:  2009-12

9.  Monoamniotic monochorionic twins discordant for noncompaction cardiomyopathy.

Authors:  Dianna Ng; Yosr Bouhlal; Philip C Ursell; Joseph T C Shieh
Journal:  Am J Med Genet A       Date:  2013-05-01       Impact factor: 2.802

10.  A mutant HCN4 channel in a family with bradycardia, left bundle branch block, and left ventricular noncompaction.

Authors:  Ryosuke Yokoyama; Koshi Kinoshita; Yukiko Hata; Masayoshi Abe; Kenta Matsuoka; Keiichi Hirono; Masanobu Kano; Makoto Nakazawa; Fukiko Ichida; Naoki Nishida; Toshihide Tabata
Journal:  Heart Vessels       Date:  2018-01-18       Impact factor: 2.037

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