Rescue of neurological deficits in a mouse model for Angelman syndrome by reduction of alphaCaMKII inhibitory phosphorylation.

Abstract:

:Angelman syndrome (AS) is a severe neurological disorder characterized by mental retardation, motor dysfunction and epilepsy. We show that the molecular and cellular deficits of an AS mouse model can be rescued by introducing an additional mutation at the inhibitory phosphorylation site of alphaCaMKII. Moreover, these double mutants no longer show the behavioral deficits seen in AS mice, suggesting that these deficits are the direct result of increased inhibitory phosphorylation of alphaCaMKII.

journal_name

Nat Neurosci

journal_title

Nature neuroscience

authors

van Woerden GM,Harris KD,Hojjati MR,Gustin RM,Qiu S,de Avila Freire R,Jiang YH,Elgersma Y,Weeber EJ

doi

10.1038/nn1845

subject

Has Abstract

pub_date

2007-03-01 00:00:00

pages

280-2

issue

3

eissn

1097-6256

issn

1546-1726

pii

nn1845

journal_volume

10

pub_type

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