[article]
| Titre : |
Deficits in skilled motor and auditory learning in a rat model of Rett syndrome |
| Type de document : |
texte imprimé |
| Auteurs : |
Katherine S. ADCOCK, Auteur ; Abigail E. BLOUNT, Auteur ; Robert A. MORRISON, Auteur ; Amanda ALVAREZ-DIEPPA, Auteur ; Michael P. KILGARD, Auteur ; Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur |
| Langues : |
Anglais (eng) |
| Mots-clés : |
Animals Auditory Perception Female Learning Methyl-CpG-Binding Protein 2/genetics Mice Mice, Transgenic Rats Rett Syndrome/complications/genetics Auditory MeCP2 Motor Rett syndrome |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Rett syndrome is an X-linked neurodevelopmental disorder caused by a mutation in the gene MECP2. Individuals with Rett syndrome display developmental regression at an early age, and develop a range of motor, auditory, cognitive, and social impairments. Several studies have successfully modeled some aspects of dysfunction and Rett syndrome-like phenotypes in transgenic mouse and rat models bearing mutations in the MECP2 gene. Here, we sought to extend these findings and characterize skilled learning, a more complex behavior known to be altered in Rett syndrome. METHODS: We evaluated the acquisition and performance of auditory and motor function on two complex tasks in heterozygous female Mecp2 rats. Animals were trained to perform a speech discrimination task or a skilled forelimb reaching task. RESULTS: Our results reveal that Mecp2 rats display slower acquisition and reduced performance on an auditory discrimination task than wild-type (WT) littermates. Similarly, Mecp2 rats exhibit impaired learning rates and worse performance on a skilled forelimb motor task compared to WT. CONCLUSIONS: Together, these findings illustrate novel deficits in skilled learning consistent with clinical manifestation of Rett syndrome and provide a framework for development of therapeutic strategies to improve these complex behaviors. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-020-09330-5 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 |
in Journal of Neurodevelopmental Disorders > 12 (2020)
[article] Deficits in skilled motor and auditory learning in a rat model of Rett syndrome [texte imprimé] / Katherine S. ADCOCK, Auteur ; Abigail E. BLOUNT, Auteur ; Robert A. MORRISON, Auteur ; Amanda ALVAREZ-DIEPPA, Auteur ; Michael P. KILGARD, Auteur ; Crystal T. ENGINEER, Auteur ; Seth A. HAYS, Auteur. Langues : Anglais ( eng) in Journal of Neurodevelopmental Disorders > 12 (2020)
| Mots-clés : |
Animals Auditory Perception Female Learning Methyl-CpG-Binding Protein 2/genetics Mice Mice, Transgenic Rats Rett Syndrome/complications/genetics Auditory MeCP2 Motor Rett syndrome |
| Index. décimale : |
PER Périodiques |
| Résumé : |
BACKGROUND: Rett syndrome is an X-linked neurodevelopmental disorder caused by a mutation in the gene MECP2. Individuals with Rett syndrome display developmental regression at an early age, and develop a range of motor, auditory, cognitive, and social impairments. Several studies have successfully modeled some aspects of dysfunction and Rett syndrome-like phenotypes in transgenic mouse and rat models bearing mutations in the MECP2 gene. Here, we sought to extend these findings and characterize skilled learning, a more complex behavior known to be altered in Rett syndrome. METHODS: We evaluated the acquisition and performance of auditory and motor function on two complex tasks in heterozygous female Mecp2 rats. Animals were trained to perform a speech discrimination task or a skilled forelimb reaching task. RESULTS: Our results reveal that Mecp2 rats display slower acquisition and reduced performance on an auditory discrimination task than wild-type (WT) littermates. Similarly, Mecp2 rats exhibit impaired learning rates and worse performance on a skilled forelimb motor task compared to WT. CONCLUSIONS: Together, these findings illustrate novel deficits in skilled learning consistent with clinical manifestation of Rett syndrome and provide a framework for development of therapeutic strategies to improve these complex behaviors. |
| En ligne : |
https://dx.doi.org/10.1186/s11689-020-09330-5 |
| Permalink : |
https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 |
|  |
Deficits in skilled motor and auditory learning in a rat model of Rett syndrome in Journal of Neurodevelopmental Disorders, 12 (2020)
