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Publication : Motor deficits and hyperactivity in Dyt1 knockdown mice.

First Author  Dang MT Year  2006
Journal  Neurosci Res Volume  56
Issue  4 Pages  470-4
PubMed ID  17046090 Mgi Jnum  J:131080
Mgi Id  MGI:3772926 Doi  10.1016/j.neures.2006.09.005
Citation  Dang MT, et al. (2006) Motor deficits and hyperactivity in Dyt1 knockdown mice. Neurosci Res 56(4):470-4
abstractText  The DYT1 gene containing a trinucleotide deletion (DeltaGAG) is linked to early-onset dystonia, a neurological movement disorder of involuntary muscle contractions. To understand DYT1's contribution to dystonia, we produced and analyzed Dyt1 knockdown (KD) mice that expressed a reduced level of torsinA protein encoded by Dyt1. Knockdown mice exhibited deficits in motor control and a decreased trend in dopamine with a significant reduction in 3,4-dihydroxyphenylacetic acid. These alterations are similar to those displayed by previously reported Dyt1 DeltaGAG knockin heterozygous mice, suggesting that the partial loss of torsinA function contributes to the pathology of the disease.
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