Dissociating motor impairment from five-choice serial reaction time task performance in a mouse model of Angelman syndrome.

ABSTRACT: Angelman syndrome (AS) is a single-gene neurodevelopmental disorder associated with cognitive and motor impairment, seizures, lack of speech, and disrupted sleep. AS is caused by loss-of-function mutations in the UBE3A gene, and approaches to reinstate functional UBE3A are currently in clinical trials in children. Behavioral testing in a mouse model of AS (Ube3a ^m-/p+ ) represents an important tool to assess the effectiveness of current and future treatments preclinically. Existing behavioral tests effectively model motor impairments, but not cognitive impairments, in Ube3a ^m-/p+ mice. Here we tested the hypothesis that the 5-choice serial reaction time task (5CSRTT) can be used to assess cognitive behaviors in Ube3a

SUBMITTER: Negron-Moreno PN 

PROVIDER: S-EPMC9539753 | biostudies-literature | 2022

REPOSITORIES: biostudies-literature