Deficits in skilled motor and auditory learning in a rat model of Rett syndrome

Journal of Neurodevelopmental Disorders

卷:12

Katherine S. Adcock¹  Amanda Alvarez-Dieppa¹  Michael P. Kilgard¹  Crystal T. Engineer¹  Seth A. Hays¹  Robert A. Morrison¹  Abigail E. Blount² 

[1] School of Behavioral and Brain Sciences, The University of Texas at Dallas;

[2] Texas Biomedical Device Center, The University of Texas at Dallas;

关键词: Rett syndrome;    MeCP2;    Motor;    Auditory;    Learning;   

DOI  :  10.1186/s11689-020-09330-5

来源: DOAJ

【 摘 要 】

Abstract 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.

【 授权许可】

Unknown