PMID- 17046090
OWN - NLM
STAT- MEDLINE
DCOM- 20070117
LR  - 20181113
IS  - 0168-0102 (Print)
IS  - 0168-0102 (Linking)
VI  - 56
IP  - 4
DP  - 2006 Dec
TI  - Motor deficits and hyperactivity in Dyt1 knockdown mice.
PG  - 470-4
AB  - 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.
FAU - Dang, Mai T
AU  - Dang MT
AD  - Department of Molecular and Integrative Physiology, NeuroTech Group, Beckman 
      Institute for Advanced Science and Technology, University of Illinois at 
      Urbana-Champaign, Urbana, IL 61801, USA.
FAU - Yokoi, Fumiaki
AU  - Yokoi F
FAU - Pence, Morgan A
AU  - Pence MA
FAU - Li, Yuqing
AU  - Li Y
LA  - eng
GR  - R21 NS047692/NS/NINDS NIH HHS/United States
GR  - NS047692/NS/NINDS NIH HHS/United States
PT  - Journal Article
PT  - Research Support, N.I.H., Extramural
PT  - Research Support, Non-U.S. Gov't
DEP - 20061013
PL  - Ireland
TA  - Neurosci Res
JT  - Neuroscience research
JID - 8500749
RN  - 0 (Dyt1 protein, mouse)
RN  - 0 (Molecular Chaperones)
RN  - 0 (RNA, Messenger)
RN  - 102-32-9 (3,4-Dihydroxyphenylacetic Acid)
RN  - 63231-63-0 (RNA)
RN  - EC 1.4.3.4 (Monoamine Oxidase)
RN  - VTD58H1Z2X (Dopamine)
RN  - X77S6GMS36 (Homovanillic Acid)
SB  - IM
MH  - 3,4-Dihydroxyphenylacetic Acid/metabolism
MH  - Alleles
MH  - Animals
MH  - Blotting, Western
MH  - Dopamine/metabolism
MH  - Exons/genetics
MH  - Genetic Vectors
MH  - Genotype
MH  - Homovanillic Acid
MH  - Hyperkinesis/*genetics/*physiopathology
MH  - Mice
MH  - Mice, Knockout
MH  - Molecular Chaperones/*genetics/*physiology
MH  - Monoamine Oxidase/metabolism
MH  - Motor Activity/physiology
MH  - Neostriatum/metabolism
MH  - Neuromuscular Diseases/*genetics/physiopathology
MH  - RNA/biosynthesis
MH  - RNA, Messenger/biosynthesis/genetics
MH  - Sex Characteristics
PMC - PMC1794321
MID - NIHMS14570
EDAT- 2006/10/19 09:00
MHDA- 2007/01/18 09:00
PMCR- 2008/01/31
CRDT- 2006/10/19 09:00
PHST- 2006/06/28 00:00 [received]
PHST- 2006/08/25 00:00 [revised]
PHST- 2006/09/08 00:00 [accepted]
PHST- 2006/10/19 09:00 [pubmed]
PHST- 2007/01/18 09:00 [medline]
PHST- 2006/10/19 09:00 [entrez]
PHST- 2008/01/31 00:00 [pmc-release]
AID - S0168-0102(06)00230-6 [pii]
AID - 10.1016/j.neures.2006.09.005 [doi]
PST - ppublish
SO  - Neurosci Res. 2006 Dec;56(4):470-4. doi: 10.1016/j.neures.2006.09.005. Epub 2006 
      Oct 13.