Literature DB >> 16687615

Impaired motor function in mice with cell-specific knockout of sodium channel Scn8a (NaV1.6) in cerebellar purkinje neurons and granule cells.

Stephen I Levin1, Zayd M Khaliq, Teresa K Aman, Tina M Grieco, Jennifer A Kearney, Indira M Raman, Miriam H Meisler.   

Abstract

The Scn8a gene encodes the voltage-gated Na channel alpha subunit Na(V)1.6, which is widely expressed throughout the nervous system. Global null mutations that eliminate Scn8a in all cells result in severe motor dysfunction and premature death, precluding analysis of the physiological role of Na(V)1.6 in different neuronal types. To test the effect of cerebellar Na(V)1.6 on motor coordination in mice, we used the Cre-lox system to eliminate Scn8a expression exclusively in Purkinje neurons (Purkinje KO) and/or granule neurons (granule KO). Whereas granule KO mice had only minor behavioral defects, adult Purkinje KO mice exhibited ataxia, tremor, and impaired coordination. These disorders were exacerbated in double mutants lacking Scn8a in both Purkinje and granule cells (double KO). In Purkinje cells isolated from adult Purkinje KO and double KO but not granule KO mice, the ratio of resurgent-to-transient tetrodotoxin- (TTX)-sensitive Na current amplitudes decreased from approximately 15 to approximately 5%. In cerebellar slices, Purkinje cell spontaneous and maximal firing rates were reduced 10-fold and twofold relative to control in Purkinje KO and double KO but not granule KO mice. Additionally, short-term plasticity of high-frequency parallel fiber EPSCs was altered relative to control in Purkinje KO and double KO but not granule KO mice. These data suggest that the specialized kinetics of Purkinje Na channels depend directly on Scn8a expression. The loss of these channels leads to a decrease in Purkinje cell firing rates as well as a modification of the synaptic properties of afferent parallel fibers, with the ultimate consequence of disrupting motor behavior.

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Year:  2006        PMID: 16687615     DOI: 10.1152/jn.01193.2005

Source DB:  PubMed          Journal:  J Neurophysiol        ISSN: 0022-3077            Impact factor:   2.714


  64 in total

1.  Loss of beta-III spectrin leads to Purkinje cell dysfunction recapitulating the behavior and neuropathology of spinocerebellar ataxia type 5 in humans.

Authors:  Emma M Perkins; Yvonne L Clarkson; Nancy Sabatier; David M Longhurst; Christopher P Millward; Jennifer Jack; Junko Toraiwa; Mitsunori Watanabe; Jeffrey D Rothstein; Alastair R Lyndon; David J A Wyllie; Mayank B Dutia; Mandy Jackson
Journal:  J Neurosci       Date:  2010-04-07       Impact factor: 6.167

2.  Cross-species conservation of open-channel block by Na channel β4 peptides reveals structural features required for resurgent Na current.

Authors:  Amanda H Lewis; Indira M Raman
Journal:  J Neurosci       Date:  2011-08-10       Impact factor: 6.167

3.  Axonal Na+ channels ensure fast spike activation and back-propagation in cerebellar granule cells.

Authors:  Shyam Diwakar; Jacopo Magistretti; Mitchell Goldfarb; Giovanni Naldi; Egidio D'Angelo
Journal:  J Neurophysiol       Date:  2008-12-10       Impact factor: 2.714

4.  Control of voluntary and optogenetically perturbed locomotion by spike rate and timing of neurons of the mouse cerebellar nuclei.

Authors:  Rashmi Sarnaik; Indira M Raman
Journal:  Elife       Date:  2018-04-16       Impact factor: 8.140

5.  Persistent Nav1.6 current at axon initial segments tunes spike timing of cerebellar granule cells.

Authors:  Nancy Osorio; Laurence Cathala; Miriam H Meisler; Marcel Crest; Jacopo Magistretti; Patrick Delmas
Journal:  J Physiol       Date:  2010-02-15       Impact factor: 5.182

6.  Biophysical characterisation of the persistent sodium current of the Nav1.6 neuronal sodium channel: a single-channel analysis.

Authors:  Aurélien Chatelier; Juan Zhao; Patrick Bois; Mohamed Chahine
Journal:  Pflugers Arch       Date:  2010-03-05       Impact factor: 3.657

7.  Delayed postnatal loss of P/Q-type calcium channels recapitulates the absence epilepsy, dyskinesia, and ataxia phenotypes of genomic Cacna1a mutations.

Authors:  Melanie D Mark; Takashi Maejima; Denise Kuckelsberg; Jong W Yoo; Robert A Hyde; Viral Shah; Davina Gutierrez; Rosa L Moreno; Wolfgang Kruse; Jeffrey L Noebels; Stefan Herlitze
Journal:  J Neurosci       Date:  2011-03-16       Impact factor: 6.167

Review 8.  Nothing can be coincidence: synaptic inhibition and plasticity in the cerebellar nuclei.

Authors:  Jason R Pugh; Indira M Raman
Journal:  Trends Neurosci       Date:  2009-01-27       Impact factor: 13.837

9.  Iberiotoxin-sensitive and -insensitive BK currents in Purkinje neuron somata.

Authors:  Mark D Benton; Amanda H Lewis; Jason S Bant; Indira M Raman
Journal:  J Neurophysiol       Date:  2013-02-27       Impact factor: 2.714

10.  Heterozygous mutations of the voltage-gated sodium channel SCN8A are associated with spike-wave discharges and absence epilepsy in mice.

Authors:  Ligia A Papale; Barbara Beyer; Julie M Jones; Lisa M Sharkey; Sergio Tufik; Michael Epstein; Verity A Letts; Miriam H Meisler; Wayne N Frankel; Andrew Escayg
Journal:  Hum Mol Genet       Date:  2009-03-02       Impact factor: 6.150

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