Literature DB >> 11150514

Biophysical phenotypes of SCN5A mutations causing long QT and Brugada syndromes.

G Baroudi1, M Chahine.   

Abstract

Long QT and Brugada syndromes are two hereditary cardiac diseases. Brugada syndrome has so far been associated with only one gene, SCN5A, which encodes the cardiac sodium channel. However, in long QT syndrome (LQTS) at least six genes, including the SCN5A, are implicated. The substitution (D1790G) causes LQTS and the insertion (D1795) induces both LQTS and Brugada syndromes in carrier patients. hH1/insD1795 and hH1/D1790G mutant channels were expressed in the tsA201 human cell line and characterized using the patch clamp technique in whole-cell configuration. Our data revealed a persistent inward sodium current of about 6% at -30 mV for both D1790G and insD1795, and a reduction of 62% of channel expression for the insD1795. Moreover, a shift of steady-state inactivation curve in both mutants was also observed. Our findings uphold the idea that LQT3 is related to a persistent sodium current whereas reduction in the expression level of cardiac sodium channels is one of the biophysical characteristics of Brugada syndrome.

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Year:  2000        PMID: 11150514     DOI: 10.1016/s0014-5793(00)02360-7

Source DB:  PubMed          Journal:  FEBS Lett        ISSN: 0014-5793            Impact factor:   4.124


  22 in total

1.  Role of the C-terminal domain in inactivation of brain and cardiac sodium channels.

Authors:  M Mantegazza; F H Yu; W A Catterall; T Scheuer
Journal:  Proc Natl Acad Sci U S A       Date:  2001-12-11       Impact factor: 11.205

2.  Structural effects of an LQT-3 mutation on heart Na+ channel gating.

Authors:  M Tateyama; H Liu; A-S Yang; J W Cormier; R S Kass
Journal:  Biophys J       Date:  2004-03       Impact factor: 4.033

3.  Regulation of persistent Na current by interactions between beta subunits of voltage-gated Na channels.

Authors:  Teresa K Aman; Tina M Grieco-Calub; Chunling Chen; Raffaella Rusconi; Emily A Slat; Lori L Isom; Indira M Raman
Journal:  J Neurosci       Date:  2009-02-18       Impact factor: 6.167

4.  Subepicardial phase 0 block and discontinuous transmural conduction underlie right precordial ST-segment elevation by a SCN5A loss-of-function mutation.

Authors:  Markéta Bébarová; Tom O'Hara; Jan L M C Geelen; Roselie J Jongbloed; Carl Timmermans; Yvonne H Arens; Luz-Maria Rodriguez; Yoram Rudy; Paul G A Volders
Journal:  Am J Physiol Heart Circ Physiol       Date:  2008-05-02       Impact factor: 4.733

5.  Electrophysiological characterization of a large set of novel variants in the SCN5A-gene: identification of novel LQTS3 and BrS mutations.

Authors:  Beatriz Ortiz-Bonnin; Susanne Rinné; Robin Moss; Anne K Streit; Michael Scharf; Katrin Richter; Anika Stöber; Arne Pfeufer; Gunnar Seemann; Stefan Kääb; Britt-Maria Beckmann; Niels Decher
Journal:  Pflugers Arch       Date:  2016-06-11       Impact factor: 3.657

6.  A C-terminal skeletal muscle sodium channel mutation associated with myotonia disrupts fast inactivation.

Authors:  Fen-fen Wu; Erynn Gordon; Eric P Hoffman; Stephen C Cannon
Journal:  J Physiol       Date:  2005-03-17       Impact factor: 5.182

7.  Functional Interactions between Distinct Sodium Channel Cytoplasmic Domains through the Action of Calmodulin.

Authors:  Franck Potet; Benjamin Chagot; Mircea Anghelescu; Prakash C Viswanathan; Svetlana Z Stepanovic; Sabina Kupershmidt; Walter J Chazin; Jeffrey R Balser
Journal:  J Biol Chem       Date:  2009-01-26       Impact factor: 5.157

8.  SCN5A (NaV1.5) Variant Functional Perturbation and Clinical Presentation: Variants of a Certain Significance.

Authors:  Brett M Kroncke; Andrew M Glazer; Derek K Smith; Jeffrey D Blume; Dan M Roden
Journal:  Circ Genom Precis Med       Date:  2018-05

9.  Atrial arrhythmogenicity in aged Scn5a+/DeltaKPQ mice modeling long QT type 3 syndrome and its relationship to Na+ channel expression and cardiac conduction.

Authors:  Laila Guzadhur; Sarah M Pearcey; Rudolf M Duehmke; Kamalan Jeevaratnam; Anja F Hohmann; Yanmin Zhang; Andrew A Grace; Ming Lei; Christopher L-H Huang
Journal:  Pflugers Arch       Date:  2010-06-16       Impact factor: 3.657

Review 10.  The cardiac persistent sodium current: an appealing therapeutic target?

Authors:  D A Saint
Journal:  Br J Pharmacol       Date:  2007-12-10       Impact factor: 8.739

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