| Budget Amount *help |
¥4,680,000 (Direct Cost: ¥3,600,000、Indirect Cost: ¥1,080,000)
Fiscal Year 2011: ¥1,300,000 (Direct Cost: ¥1,000,000、Indirect Cost: ¥300,000)
Fiscal Year 2010: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
Fiscal Year 2009: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000)
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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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