Literature DB >> 24682880

Enhanced slow inactivation of the human skeletal muscle sodium channel causing normokalemic periodic paralysis.

Lei Wu1, Baorong Zhang, Ying Kang, Weiping Wu.   

Abstract

Normokalemic periodic paralysis (normoPP) is a type of skeletal muscle function disorder which is characterized by paralysis attack with concomitant normal serum potassium level. We previously reported that R675Q mutation of human skeletal muscle voltage-gated sodium channel α subunit (SCN4A) may be the novel mutation which caused normoPP in Chinese families. However, it is still not clear how this mutation affects the SCN4A channel function. In this study, we used patch-clamp recording to study the function of wild type (WT) and R675Q mutant of SCN4A channels expressed in human embryonic kidney (HEK293) cells. We found that R675Q mutation did not affect the voltage dependence of sodium channel activation. The fast inactivation was also not significantly affected by R675Q mutation. However, R675Q mutation of SCN4A channels exhibited an 11.1 mV hyperpolarized shift in the voltage dependence of slow inactivation and significantly prolonged the recovery from prolonged inactivation state. Our results thus indicate that SCN4A was functionally affected by R675Q mutation, suggesting a possible reason for causing normoPP in Chinese patients.

Entities:  

Mesh:

Substances:

Year:  2014        PMID: 24682880     DOI: 10.1007/s10571-014-0052-y

Source DB:  PubMed          Journal:  Cell Mol Neurobiol        ISSN: 0272-4340            Impact factor:   5.046


  30 in total

1.  Slow inactivation differs among mutant Na channels associated with myotonia and periodic paralysis.

Authors:  L J Hayward; R H Brown; S C Cannon
Journal:  Biophys J       Date:  1997-03       Impact factor: 4.033

Review 2.  Troubleshooting problems with in vitro screening of drugs for QT interval prolongation using HERG K+ channels expressed in mammalian cell lines and Xenopus oocytes.

Authors:  Harry J Witchel; James T Milnes; John S Mitcheson; Jules C Hancox
Journal:  J Pharmacol Toxicol Methods       Date:  2002 Sep-Oct       Impact factor: 1.950

3.  Amino acid residues required for fast Na(+)-channel inactivation: charge neutralizations and deletions in the III-IV linker.

Authors:  D E Patton; J W West; W A Catterall; A L Goldin
Journal:  Proc Natl Acad Sci U S A       Date:  1992-11-15       Impact factor: 11.205

4.  Normokalemic periodic paralysis revisited: does it exist?

Authors:  Patrick F Chinnery; Timothy J Walls; Michael G Hanna; David Bates; Peter R W Fawcett
Journal:  Ann Neurol       Date:  2002-08       Impact factor: 10.422

5.  Impaired slow inactivation in mutant sodium channels.

Authors:  T R Cummins; F J Sigworth
Journal:  Biophys J       Date:  1996-07       Impact factor: 4.033

6.  Enhanced inactivation and pH sensitivity of Na(+) channel mutations causing hypokalaemic periodic paralysis type II.

Authors:  Alexey Kuzmenkin; Vanesa Muncan; Karin Jurkat-Rott; Chao Hang; Holger Lerche; Frank Lehmann-Horn; Nenad Mitrovic
Journal:  Brain       Date:  2002-04       Impact factor: 13.501

7.  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

8.  Depolarization-activated gating pore current conducted by mutant sodium channels in potassium-sensitive normokalemic periodic paralysis.

Authors:  Stanislav Sokolov; Todd Scheuer; William A Catterall
Journal:  Proc Natl Acad Sci U S A       Date:  2008-12-03       Impact factor: 11.205

9.  Sodium channel inactivation is altered by substitution of voltage sensor positive charges.

Authors:  K J Kontis; A L Goldin
Journal:  J Gen Physiol       Date:  1997-10       Impact factor: 4.086

10.  Mechanisms of a human skeletal myotonia produced by mutation in the C-terminus of NaV1.4: is Ca2+ regulation defective?

Authors:  Subrata Biswas; Deborah A DiSilvestre; Peihong Dong; Gordon F Tomaselli
Journal:  PLoS One       Date:  2013-12-06       Impact factor: 3.240

View more
  4 in total

1.  SCN4A p.R675Q Mutation Leading to Normokalemic Periodic Paralysis: A Family Report and Literature Review.

Authors:  Jiejing Shi; Qianqian Qu; Haiyan Liu; Wenhao Cui; Yan Zhang; Haidong Lv; Zuneng Lu
Journal:  Front Neurol       Date:  2019-10-25       Impact factor: 4.003

2.  A recessive Nav1.4 mutation underlies congenital myasthenic syndrome with periodic paralysis.

Authors:  Karima Habbout; Hugo Poulin; François Rivier; Serena Giuliano; Damien Sternberg; Bertrand Fontaine; Bruno Eymard; Raul Juntas Morales; Bernard Echenne; Louise King; Michael G Hanna; Roope Männikkö; Mohamed Chahine; Sophie Nicole; Said Bendahhou
Journal:  Neurology       Date:  2015-12-11       Impact factor: 9.910

Review 3.  Physiological and Pathophysiological Insights of Nav1.4 and Nav1.5 Comparison.

Authors:  Gildas Loussouarn; Damien Sternberg; Sophie Nicole; Céline Marionneau; Francoise Le Bouffant; Gilles Toumaniantz; Julien Barc; Olfat A Malak; Véronique Fressart; Yann Péréon; Isabelle Baró; Flavien Charpentier
Journal:  Front Pharmacol       Date:  2016-01-14       Impact factor: 5.810

4.  Familial Normokalemic Periodic Paralysis Associated With Mutation in the SCN4A p.M1592V.

Authors:  Chao Fu; Zhenyu Wang; Libo Wang; Jia Li; Qiuling Sang; Jiajun Chen; Ling Qi; Hui Jin; Xiaoyang Liu
Journal:  Front Neurol       Date:  2018-06-07       Impact factor: 4.003

  4 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.