Literature DB >> 10200053

A de novo missense mutation (R1623Q) of the SCN5A gene in a Japanese girl with sporadic long QT sydrome. Mutations in brief no. 140. Online.

H Yamagishi1, M Furutani, M Kamisago, Y Morikawa, Y Kojima, Y Hino, Y Furutani, M Kimura, S Imamura, A Takao, K Momma, R Matsuoka.   

Abstract

Two missense mutations and a nine-nucleotide deletion of the cardiac sodium channel (SCN5A) gene have been shown to cause long QT syndrome (LQTS) in several familial cases. We identified a novel missense mutation (R1623Q) of the SCN5A gene in a Japanese girl with sporadic LQTS. We used polymerase chain reaction, single-strand conformation polymorphism analysis and DNA sequence analysis to identify a mutation of the SCN5A gene in the patient. A single nucleotide substitution of guanine to adenine, in codon 1612, changed the coding sense of the SCN5A from arginine to glutamine (R1623Q) in the S4 segment of domain IV which is a highly conserved region of the SCN5A. This mutation was not identified in the unaffected biological parents and brother of the patient, and 100 normal, unrelated individuals. This finding is the first evidence of a de nova mutation in SCN5A associated with LQTS.

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Year:  1998        PMID: 10200053     DOI: 10.1002/(SICI)1098-1004(1998)11:6<481::AID-HUMU12>3.0.CO;2-Q

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


  6 in total

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2.  Developmentally regulated SCN5A splice variant potentiates dysfunction of a novel mutation associated with severe fetal arrhythmia.

Authors:  Lisa L Murphy; Anita J Moon-Grady; Bettina F Cuneo; Ronald T Wakai; Suhong Yu; Jennifer D Kunic; D Woodrow Benson; Alfred L George
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3.  Normalization of ventricular repolarization with flecainide in long QT syndrome patients with SCN5A:DeltaKPQ mutation.

Authors:  J R Windle; R C Geletka; A J Moss; W Zareba; D L Atkins
Journal:  Ann Noninvasive Electrocardiol       Date:  2001-04       Impact factor: 1.468

4.  Malignant perinatal variant of long-QT syndrome caused by a profoundly dysfunctional cardiac sodium channel.

Authors:  Dao W Wang; Lia Crotti; Wataru Shimizu; Matteo Pedrazzini; Francesco Cantu; Paolo De Filippo; Kanako Kishiki; Aya Miyazaki; Tomoaki Ikeda; Peter J Schwartz; Alfred L George
Journal:  Circ Arrhythm Electrophysiol       Date:  2008-12-02

5.  Pathophysiological role of omega pore current in channelopathies.

Authors:  Karin Jurkat-Rott; James Groome; Frank Lehmann-Horn
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6.  The diagnosis and management of long QT syndrome based on fetal echocardiography.

Authors:  Benjamin A Blais; Gary Satou; Mark S Sklansky; Himani Madnawat; Jeremy P Moore
Journal:  HeartRhythm Case Rep       Date:  2017-07-18
  6 in total

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