Paroxysmal Dyskinesias in Mice
Identifieur interne : 000020 ( Pmc/Curation ); précédent : 000019; suivant : 000021Paroxysmal Dyskinesias in Mice
Auteurs : Thomas L. Shirley ; Lekha M. Rao ; Ellen J. Hess ; H. A. JinnahSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2008.
Abstract
Animal models of human disease are important tools for revealing the underlying mechanisms of pathophysiology and developing therapeutic strategies. Several unique mouse calcium channel mutants have been identified with nonepileptic, episodic dyskinetic movements that are phenotypically similar to human paroxysmal dyskinesias. In this report, video demonstrations of these motor attacks are provided for two previously described mouse mutants, tottering and lethargic, as well as a new one, rocker. Semiquantitative comparisons using two different rating scales reveal differences in attack morphology, severity, and duration among the strains. These mice provide three independent models of paroxysmal dyskinesia and support for prior proposals that channelopathies may underlie the human disorders.
Url:
DOI: 10.1002/mds.21829
PubMed: 17999434
PubMed Central: 2887756
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PMC:2887756Le document en format XML
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<author><name sortKey="Rao, Lekha M" sort="Rao, Lekha M" uniqKey="Rao L" first="Lekha M." last="Rao">Lekha M. Rao</name>
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<author><name sortKey="Hess, Ellen J" sort="Hess, Ellen J" uniqKey="Hess E" first="Ellen J." last="Hess">Ellen J. Hess</name>
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<author><name sortKey="Jinnah, H A" sort="Jinnah, H A" uniqKey="Jinnah H" first="H. A." last="Jinnah">H. A. Jinnah</name>
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<front><div type="abstract" xml:lang="en"><p id="P1">Animal models of human disease are important tools for revealing the underlying mechanisms of pathophysiology and developing therapeutic strategies. Several unique mouse calcium channel mutants have been identified with nonepileptic, episodic dyskinetic movements that are phenotypically similar to human paroxysmal dyskinesias. In this report, video demonstrations of these motor attacks are provided for two previously described mouse mutants, tottering and lethargic, as well as a new one, rocker. Semiquantitative comparisons using two different rating scales reveal differences in attack morphology, severity, and duration among the strains. These mice provide three independent models of paroxysmal dyskinesia and support for prior proposals that channelopathies may underlie the human disorders.</p>
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<contrib-group><contrib contrib-type="author"><name><surname>Shirley</surname>
<given-names>Thomas L.</given-names>
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<degrees>PhD</degrees>
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<contrib contrib-type="author"><name><surname>Rao</surname>
<given-names>Lekha M.</given-names>
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<contrib contrib-type="author"><name><surname>Hess</surname>
<given-names>Ellen J.</given-names>
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<degrees>PhD</degrees>
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<contrib contrib-type="author"><name><surname>Jinnah</surname>
<given-names>H. A.</given-names>
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<degrees>MD, PhD</degrees>
<xref rid="FN1" ref-type="author-notes">*</xref>
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<aff id="A1">Department of Neurology, Johns Hopkins University, Baltimore, Maryland, USA</aff>
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<author-notes><corresp id="FN1"><label>*</label>
Correspondence to: H.A. Jinnah, Meyer 6-181, Johns Hopkins University, Baltimore, MD 21287. E-mail: <email>hjinnah@jhmi.edu</email>
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<pub-date pub-type="nihms-submitted"><day>17</day>
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<year>2008</year>
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<pub-date pub-type="pmc-release"><day>18</day>
<month>6</month>
<year>2010</year>
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<volume>23</volume>
<issue>2</issue>
<fpage>259</fpage>
<lpage>264</lpage>
<abstract><p id="P1">Animal models of human disease are important tools for revealing the underlying mechanisms of pathophysiology and developing therapeutic strategies. Several unique mouse calcium channel mutants have been identified with nonepileptic, episodic dyskinetic movements that are phenotypically similar to human paroxysmal dyskinesias. In this report, video demonstrations of these motor attacks are provided for two previously described mouse mutants, tottering and lethargic, as well as a new one, rocker. Semiquantitative comparisons using two different rating scales reveal differences in attack morphology, severity, and duration among the strains. These mice provide three independent models of paroxysmal dyskinesia and support for prior proposals that channelopathies may underlie the human disorders.</p>
</abstract>
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<contract-sponsor id="NS1">National Institute of Neurological Disorders and Stroke : NINDS</contract-sponsor>
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