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Updated: Jul 17, 2026
Behavioral Characterization of an Angelman Syndrome Mouse Model
Published on: October 20, 2023
Rescue of neurological deficits in a mouse model for Angelman syndrome by reduction of alphaCaMKII inhibitory
Geeske M van Woerden1, Karen D Harris, Mohammad Reza Hojjati
1Erasmus MC, University Medical Centre, Department of Neuroscience, Dr. Molewaterplein 50, PO Box 2040, 3000 CA, Rotterdam, The Netherlands.
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.
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