2011 Fiscal Year Final Research Report
Physiological function of Na pumpα3 subunit gene and involvement in pathophysiology of dystonia parkinsonism.
| Project/Area Number |
21590239
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| Research Category |
Grant-in-Aid for Scientific Research (C)
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| Allocation Type | Single-year Grants |
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| Section | 一般 |
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| Research Field |
General physiology
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| Research Institution | Jichi Medical University |
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Principal Investigator |
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| Co-Investigator(Renkei-kenkyūsha) |
IKEDA Keiko 兵庫医科大学, 医学部, 主任教授 (10265241)
ONAKA Tatsushi 自治医科大学, 医学部, 教授 (90177254)
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| Project Period (FY) |
2009 – 2011
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| Keywords | ナトリウムポンプα3サブユニット / 小脳 / プルキンエ細胞 / シナプス伝達 / 電気生理学 / 抑制性ニューロン |
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| Research Abstract |
Dystonia is characterized by excessive involuntary and prolonged simultaneous contractions of both agonist and antagonist muscles. One hereditary form of dystonia, DYT12, is caused by loss of function mutations of the gene forα3 isoform of Na pump(ATP1A3). To provide insight into the molecular etiology of DYT12, we generated and analyzed knockout heterozygous mice(Atp1a3^<+/->). Atp1a3^<+/-> mice showed increased sensitivity to kainate-induced dystonia than wild-type littermates(WT). Electrophysiological studies showed enhanced response of Purkinje cells in Atp1a3^<+/-> at cerebellar inhibitory synapses. Our results shed light on the role of Atp1a3 in inhibitory synapse, and its potential involvement in the expression of dystonia symptoms of DYT12.
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