Literature DB >> 19225109

K+-dependent paradoxical membrane depolarization and Na+ overload, major and reversible contributors to weakness by ion channel leaks.

Karin Jurkat-Rott1, Marc-André Weber, Michael Fauler, Xiu-Hai Guo, Boris D Holzherr, Agathe Paczulla, Nikolai Nordsborg, Wolfgang Joechle, Frank Lehmann-Horn.   

Abstract

Normal resting potential (P1) of myofibers follows the Nernst equation, exhibiting about -85 mV at a normal extracellular K(+) concentration ([K(+)](o)) of 4 mM. Hyperpolarization occurs with decreased [K(+)](o), although at [K(+)](o) < 1.0 mM, myofibers paradoxically depolarize to a second stable potential of -60 mV (P2). In rat myofiber bundles, P2 also was found at more physiological [K(+)](o) and was associated with inexcitability. To increase the relative frequency of P2 to 50%, [K(+)](o) needed to be lowered to 1.5 mM. In the presence of the ionophore gramicidin, [K(+)](o) reduction to only 2.5 mM yielded the same effect. Acetazolamide normalized this increased frequency of P2 fibers. The findings mimic hypokalemic periodic paralysis (HypoPP), a channelopathy characterized by hypokalemia-induced weakness. Of myofibers from 7 HypoPP patients, up to 25% were in P2 at a [K(+)](o) of 4 mM, in accordance with their permanent weakness, and up to 99% were in P2 at a [K(+)](o) of 1.5 mM, in accordance with their paralytic attacks. Of 36 HypoPP patients, 25 had permanent weakness and myoplasmic intracellular Na(+) ([Na(+)](i)) overload (up to 24 mM) as shown by in vivo (23)Na-MRI. Acetazolamide normalized [Na(+)](i) and increased muscle strength. HypoPP myofibers showed a nonselective cation leak of 12-19.5 microS/cm(2), which may explain the Na(+) overload. The leak sensitizes myofibers to reduced serum K(+), and the resulting membrane depolarization causes the weakness. We postulate that the principle of paradoxical depolarization and loss of function upon [K(+)](o) reduction may apply to other tissues, such as heart or brain, when they become leaky (e.g., because of ischemia).

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Year:  2009        PMID: 19225109      PMCID: PMC2644652          DOI: 10.1073/pnas.0811277106

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  31 in total

1.  Hypokalaemic periodic paralysis type 2 caused by mutations at codon 672 in the muscle sodium channel gene SCN4A.

Authors:  D Sternberg; T Maisonobe; K Jurkat-Rott; S Nicole; E Launay; D Chauveau; N Tabti; F Lehmann-Horn; B Hainque; B Fontaine
Journal:  Brain       Date:  2001-06       Impact factor: 13.501

2.  Insulin acts in hypokalemic periodic paralysis by reducing inward rectifier K+ current.

Authors:  R L Ruff
Journal:  Neurology       Date:  1999-10-22       Impact factor: 9.910

3.  Mutations in Kir2.1 cause the developmental and episodic electrical phenotypes of Andersen's syndrome.

Authors:  N M Plaster; R Tawil; M Tristani-Firouzi; S Canún; S Bendahhou; A Tsunoda; M R Donaldson; S T Iannaccone; E Brunt; R Barohn; J Clark; F Deymeer; A L George; F A Fish; A Hahn; A Nitu; C Ozdemir; P Serdaroglu; S H Subramony; G Wolfe; Y H Fu; L J Ptácek
Journal:  Cell       Date:  2001-05-18       Impact factor: 41.582

4.  Acetazolamide opens the muscular KCa2+ channel: a novel mechanism of action that may explain the therapeutic effect of the drug in hypokalemic periodic paralysis.

Authors:  D Tricarico; M Barbieri; D C Camerino
Journal:  Ann Neurol       Date:  2000-09       Impact factor: 10.422

5.  Aldosterone- and testosterone-mediated intracellular calcium response in skeletal muscle cell cultures.

Authors:  M Estrada; J L Liberona; M Miranda; E Jaimovich
Journal:  Am J Physiol Endocrinol Metab       Date:  2000-07       Impact factor: 4.310

6.  Voltage-sensor sodium channel mutations cause hypokalemic periodic paralysis type 2 by enhanced inactivation and reduced current.

Authors:  K Jurkat-Rott; N Mitrovic; C Hang; A Kouzmekine; P Iaizzo; J Herzog; H Lerche; S Nicole; J Vale-Santos; D Chauveau; B Fontaine; F Lehmann-Horn
Journal:  Proc Natl Acad Sci U S A       Date:  2000-08-15       Impact factor: 11.205

Review 7.  Neurological disorders caused by inherited ion-channel mutations.

Authors:  Dimitri M Kullmann; Michael G Hanna
Journal:  Lancet Neurol       Date:  2002-07       Impact factor: 44.182

Review 8.  What is the optimal serum potassium level in cardiovascular patients?

Authors:  John E Macdonald; Allan D Struthers
Journal:  J Am Coll Cardiol       Date:  2004-01-21       Impact factor: 24.094

9.  Effects of chloride transport on bistable behaviour of the membrane potential in mouse skeletal muscle.

Authors:  R J Geukes Foppen; H G J van Mil; J Siegenbeek van Heukelom
Journal:  J Physiol       Date:  2002-07-01       Impact factor: 5.182

10.  Nav1.4 deregulation in dystrophic skeletal muscle leads to Na+ overload and enhanced cell death.

Authors:  Carole Hirn; George Shapovalov; Olivier Petermann; Emmanuelle Roulet; Urs T Ruegg
Journal:  J Gen Physiol       Date:  2008-07-14       Impact factor: 4.086

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  72 in total

1.  Leaky sodium channels from voltage sensor mutations in periodic paralysis, but not paramyotonia.

Authors:  David G Francis; Volodymyr Rybalchenko; Arie Struyk; Stephen C Cannon
Journal:  Neurology       Date:  2011-04-13       Impact factor: 9.910

Review 2.  Extracellular potassium homeostasis: insights from hypokalemic periodic paralysis.

Authors:  Chih-Jen Cheng; Elizabeth Kuo; Chou-Long Huang
Journal:  Semin Nephrol       Date:  2013-05       Impact factor: 5.299

3.  A sodium channel knockin mutant (NaV1.4-R669H) mouse model of hypokalemic periodic paralysis.

Authors:  Fenfen Wu; Wentao Mi; Dennis K Burns; Yu Fu; Hillery F Gray; Arie F Struyk; Stephen C Cannon
Journal:  J Clin Invest       Date:  2011-09-01       Impact factor: 14.808

Review 4.  Voltage-sensor mutations in channelopathies of skeletal muscle.

Authors:  Stephen C Cannon
Journal:  J Physiol       Date:  2010-02-15       Impact factor: 5.182

Review 5.  A tug-of-war between the host and the pathogen generates strategic hotspots for the development of novel therapeutic interventions against infectious diseases.

Authors:  Aarti Rana; Mushtaq Ahmed; Abdur Rub; Yusuf Akhter
Journal:  Virulence       Date:  2015-06-24       Impact factor: 5.882

6.  [Muscle channelopathies. Myotonias and periodic paralyses].

Authors:  K Jurkat-Rott; H Lerche; F Lehmann-Horn
Journal:  Nervenarzt       Date:  2011-04       Impact factor: 1.214

7.  Elevated resting H+ current in the R1239H type 1 hypokalaemic periodic paralysis mutated Ca2+ channel.

Authors:  Clarisse Fuster; Jimmy Perrot; Christine Berthier; Vincent Jacquemond; Bruno Allard
Journal:  J Physiol       Date:  2017-09-24       Impact factor: 5.182

Review 8.  When muscle Ca2+ channels carry monovalent cations through gating pores: insights into the pathophysiology of type 1 hypokalaemic periodic paralysis.

Authors:  Bruno Allard; Clarisse Fuster
Journal:  J Physiol       Date:  2018-04-15       Impact factor: 5.182

Review 9.  Sodium channelopathies of skeletal muscle result from gain or loss of function.

Authors:  Karin Jurkat-Rott; Boris Holzherr; Michael Fauler; Frank Lehmann-Horn
Journal:  Pflugers Arch       Date:  2010-03-17       Impact factor: 3.657

Review 10.  Skeletal muscle channelopathies: new insights into the periodic paralyses and nondystrophic myotonias.

Authors:  Daniel Platt; Robert Griggs
Journal:  Curr Opin Neurol       Date:  2009-10       Impact factor: 5.710

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