Motor deficits and hyperactivity in Dyt1 knockdown mice.

Abstract:

: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.

journal_name

Neurosci Res

journal_title

Neuroscience research

authors

Dang MT,Yokoi F,Pence MA,Li Y

doi

10.1016/j.neures.2006.09.005

subject

Has Abstract

pub_date

2006-12-01 00:00:00

pages

470-4

issue

4

eissn

0168-0102

issn

1872-8111

pii

S0168-0102(06)00230-6

journal_volume

56

pub_type

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