Altered structural and functional synaptic plasticity with motor skill learning in a mouse model of fragile x syndrome.
Open accessAbstract
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.
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In Fragile X syndrome model animals, spine turnover increased in various cortical areas.
“In vivo studies further showed that in such animals spine turnover increased in various cortical areas (Cruz-Martin et al., 2010; Pan et al., 2010; Padmashri et al., 2013), and neither whisker trimming nor motor learning could further alter spine dynamics (Pan et al., 2010; Padmashri et al., 2013).”
From Spatiotemporal dynamics of dendritic spines in the living brain (2014) · cites this paper as Padmashri et al., 2013
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- Authors
- Padmashri, R., Reiner, B. C., Suresh, A., Spartz, E., & Dunaevsky, A.
- Affiliations
- University of Nebraska Medical Center (US)
- Venue
- The Journal of Neuroscience
- Publisher
- Society for Neuroscience
- Published
- 2013-12-11
- Volume
- 33
- Issue
- 50
- Pages
- 19715-19723
- ISSN
- 0270-6474 (ISSN-L)
- 1529-2401
- PMC
- PMC3858638
- OpenAlex
- W2032123764
- Cited by
- 96
- References
- 68
- Citations by year
- 2026: 7 · 2025: 4 · 2024: 8 · 2023: 6 · 2022: 4 · 2021: 11 · 2020: 8 · 2019: 7 · 2018: 7 · 2017: 3 · 2016: 9 · 2015: 13 · 2014: 9
- FWCI
- 6.85
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Licence
- Locations
- Journal of Neuroscience (publishedVersion, cc-by-nc-sa)
- Europe PMC (PubMed Central) (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institute of Child Health and Human Development
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (R01 HD67218, R01HD067218)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
- Life Sciences › Neuroscience › Cognitive Neuroscience › Motor Control and Adaptation
- Keywords
- fragile X syndrome, FMRP, Fmr1 knockout mice, motor skill learning, synaptic plasticity, long-term potentiation, primary motor cortex, GluR1, dendritic spines, intellectual disability
- MeSH terms
- Animals, Disease Models, Animal, Fragile X Syndrome, Learning, Motor Skills, Neuronal Plasticity, Synapses, Long-Term Potentiation, Receptors, AMPA, Mice, Knockout, Mice, Fragile X Messenger Ribonucleoprotein 1
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