Multidimensional analysis of behavior predicts genotype with high accuracy in a mouse model of Angelman syndrome.

ABSTRACT: Angelman syndrome (AS) is a neurodevelopmental disorder caused by loss of expression of the maternal copy of the UBE3A gene. Individuals with AS have a multifaceted behavioral phenotype consisting of deficits in motor function, epilepsy, cognitive impairment, sleep abnormalities, as well as other comorbidities. Effectively modeling this behavioral profile and measuring behavioral improvement will be crucial for the success of ongoing and future clinical trials. Foundational studies have defined an array of behavioral phenotypes in the AS mouse model. However, no single behavioral test is able to fully capture the complex nature of AS-in mice, or in children. We performed multidimensional analysis (principal component analysis + k-means clustering) to quantify the performance of AS model mi

SUBMITTER: Tanas JK 

PROVIDER: S-EPMC9529912 | biostudies-literature | 2022 Oct

REPOSITORIES: biostudies-literature