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• W2032123764 abstract "Fragile X syndrome (FXS) is the most common inherited intellectual disability. FXS results from a mutation that causes silencing of the FMR1 gene, which encodes the fragile X mental retardation protein. Patients with FXS exhibit a range of neurological deficits, including motor skill deficits. Here, we have investigated motor skill learning and its synaptic correlates in the fmr1 knock-out (KO) mouse. We find that fmr1 KO mice have impaired motor skill learning of a forelimb-reaching task, compared with their wild-type (WT) littermate controls. Electrophysiological recordings from the forelimb region of the primary motor cortex demonstrated reduced, training-induced synaptic strengthening in the trained hemisphere. Moreover, long-term potentiation (LTP) is impaired in the fmr1 KO mouse, and motor skill training does not occlude LTP as it does in the WT mice. Whereas motor skill training induces an increase of synaptic AMPA-type glutamate receptor subunit 1 (GluA1), there is a delay in GluA1 increase in the trained hemisphere of the fmr1 KO mice. Using transcranial in vivo multiphoton microscopy, we find that fmr1 KO mice have similar spine density but increased dendritic spine turnover compared with WT mice. Finally, we report that motor skill training-induced formation of dendritic spines is impaired in fmr1 KO mice. We conclude that FMRP plays a role in motor skill learning and that reduced functional and structural synaptic plasticity might underlie the behavioral deficit in the fmr1 KO mouse." @default.
• W2032123764 created "2016-06-24" @default.
• W2032123764 creator A5006965852 @default.
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• W2032123764 creator A5046700829 @default.
• W2032123764 creator A5065424720 @default.
• W2032123764 creator A5072499785 @default.
• W2032123764 date "2013-12-11" @default.
• W2032123764 modified "2023-10-12" @default.
• W2032123764 title "Altered Structural and Functional Synaptic Plasticity with Motor Skill Learning in a Mouse Model of Fragile X Syndrome" @default.
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• W2032123764 doi "https://doi.org/10.1523/jneurosci.2514-13.2013" @default.
• W2032123764 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/3858638" @default.
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• W2032123764 hasPublicationYear "2013" @default.
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