Home LiteratureArticle Details
PMID: 11234013 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

A sodium-channel mutation causes isolated cardiac conduction disease.

Nature ·Vol. 409 ·No. 6823 ·2001-02-22 ·Pages 1043-7

Tan HL, Bink-Boelkens MT, Bezzina CR, Viswanathan PC, Beaufort-Krol GC, van Tintelen PJ, van den Berg MP, Wilde AA, Balser JR

Abstract

Cardiac conduction disorders slow the heart rhythm and cause disability in millions of people worldwide. Inherited mutations in SCN5A, the gene encoding the human cardiac sodium (Na+) channel, have been associated with rapid heart rhythms that occur suddenly and are life-threatening; however, a chief function of the Na+ channel is to initiate cardiac impulse conduction. Here we provide the first functional characterization of an SCN5A mutation that causes a sustained, isolated conduction defect with pathological slowing of the cardiac rhythm. By analysing the SCN5A coding region, we have identified a single mutation in five affected family members; this mutation results in the substitution of cysteine 514 for glycine (G514C) in the channel protein. Biophysical characterization of the mutant channel shows that there are abnormalities in voltage-dependent 'gating' behaviour that can be partially corrected by dexamethasone, consistent with the salutary effects of glucocorticoids on the clinical phenotype. Computational analysis predicts that the gating defects of G514C selectively slow myocardial conduction, but do not provoke the rapid cardiac arrhythmias associated previously with SCN5A mutations.

MeSH Terms
Action Potentials Amino Acid Substitution Arrhythmias, Cardiac/genetics Child Child, Preschool Cysteine DNA Mutational Analysis Dexamethasone/pharmacology Female Glycine Heart Conduction System Humans Ion Channel Gating/drug effects Models, Neurological Mutation NAV1.5 Voltage-Gated Sodium Channel Polymorphism, Single-Stranded Conformational Sodium Channels/drug effects,genetics,physiology
Chemicals
NAV1.5 Voltage-Gated Sodium Channel SCN5A protein, human Sodium Channels Dexamethasone Cysteine Glycine
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Tan H L
The Experimental and Molecular Cardiology Group, Academic Medical Center, University of Amsterdam, The Netherlands.
Bink-Boelkens M T
Bezzina C R
Viswanathan P C
Beaufort-Krol G C
van Tintelen P J
van den Berg M P
Wilde A A
Balser J R
Article Info
Journal
Nature
Abbr.
Nature
ISSN
0028-0836
Published
2001-02-22
Pages
1043-7
Language
English
Region
England
NLM ID
0410462
Subset
IM
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