Literature DB >> 8961180

The equine periodic paralysis Na+ channel mutation alters molecular transitions between the open and inactivated states.

W J Hanna1, R G Tsushima, R Sah, L J McCutcheon, E Marban, P H Backx.   

Abstract

1. The Na+ channel mutation associated with equine hyperkalaemic periodic paralysis (HPP) affects a highly conserved phenylalanine residue in an unexplored region of the alpha-subunit. This mutation was introduced into the rat skeletal muscle Na+ channel gene at the corresponding location (i.e. F1412L) for functional expression and characterization in Xenopus oocytes. 2. In comparison with wild-type (WT) channels, equine HPP channels showed clear evidence for disruption of inactivation: increased time-to-peak current, slowed rates of whole-cell current decay, significant increases in sustained current, rightward shifts in the steady-state inactivation curve by 9.5 mV, a 6-fold acceleration in the rate of recovery from inactivation at -80 mV, decreased number of blank single-channel sweeps, repetitive opening of single channels throughout depolarizing steps, increased open probability per sweep, and an increased mean open time. 3. The observed disruption of inactivation in HPP occurred without measurable changes in steady-state activation and first latency kinetics of channel opening. 4. Kinetic modelling demonstrates that the equine HPP phenotype can be simulated by altering the rate constants for transitions entering and leaving the inactivated states resulting from an energetic destabilization of the inactivated state. 5. These results suggest that the highly conserved cytoplasmic end of the third transmembrane segment (S3) in the fourth internal repeat domain (domain IV) plays a critical role in Na+ channel inactivation.

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Year:  1996        PMID: 8961180      PMCID: PMC1160989          DOI: 10.1113/jphysiol.1996.sp021773

Source DB:  PubMed          Journal:  J Physiol        ISSN: 0022-3751            Impact factor:   5.182


  42 in total

1.  Restoration of inactivation in mutants of Shaker potassium channels by a peptide derived from ShB.

Authors:  W N Zagotta; T Hoshi; R W Aldrich
Journal:  Science       Date:  1990-10-26       Impact factor: 47.728

2.  Monte-Carlo-simulations of voltage fluctuations in biological membranes in the case of small numbers of transport units.

Authors:  B Kleutsch; E Freland
Journal:  Eur Biophys J       Date:  1991       Impact factor: 1.733

3.  Structural parts involved in activation and inactivation of the sodium channel.

Authors:  W Stühmer; F Conti; H Suzuki; X D Wang; M Noda; N Yahagi; H Kubo; S Numa
Journal:  Nature       Date:  1989-06-22       Impact factor: 49.962

4.  Primary structure and functional expression of a mammalian skeletal muscle sodium channel.

Authors:  J S Trimmer; S S Cooperman; S A Tomiko; J Y Zhou; S M Crean; M B Boyle; R G Kallen; Z H Sheng; R L Barchi; F J Sigworth
Journal:  Neuron       Date:  1989-07       Impact factor: 17.173

5.  Stochastic behavior of a many-channel membrane system.

Authors:  M B Jackson
Journal:  Biophys J       Date:  1985-02       Impact factor: 4.033

6.  Different effects on gating of three myotonia-causing mutations in the inactivation gate of the human muscle sodium channel.

Authors:  N Mitrović; A L George; H Lerche; S Wagner; C Fahlke; F Lehmann-Horn
Journal:  J Physiol       Date:  1995-08-15       Impact factor: 5.182

7.  Potassium channel inactivation peptide blocks cyclic nucleotide-gated channels by binding to the conserved pore domain.

Authors:  R H Kramer; E Goulding; S A Siegelbaum
Journal:  Neuron       Date:  1994-03       Impact factor: 17.173

8.  A mutation in segment IVS6 disrupts fast inactivation of sodium channels.

Authors:  J C McPhee; D S Ragsdale; T Scheuer; W A Catterall
Journal:  Proc Natl Acad Sci U S A       Date:  1994-12-06       Impact factor: 11.205

9.  Improved patch-clamp techniques for high-resolution current recording from cells and cell-free membrane patches.

Authors:  O P Hamill; A Marty; E Neher; B Sakmann; F J Sigworth
Journal:  Pflugers Arch       Date:  1981-08       Impact factor: 3.657

Review 10.  The skeletal muscle sodium and chloride channel diseases.

Authors:  A J Hudson; G C Ebers; D E Bulman
Journal:  Brain       Date:  1995-04       Impact factor: 13.501

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  2 in total

1.  Conduction through the inward rectifier potassium channel, Kir2.1, is increased by negatively charged extracellular residues.

Authors:  Nazzareno D'Avanzo; Hee Cheol Cho; Illya Tolokh; Roman Pekhletski; Igor Tolokh; Chris Gray; Saul Goldman; Peter H Backx
Journal:  J Gen Physiol       Date:  2005-04-11       Impact factor: 4.086

Review 2.  Genotype-phenotype correlation and therapeutic rationale in hyperkalemic periodic paralysis.

Authors:  Karin Jurkat-Rott; Frank Lehmann-Horn
Journal:  Neurotherapeutics       Date:  2007-04       Impact factor: 7.620

  2 in total

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