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- W2916237377 endingPage "107000" @default.
- W2916237377 startingPage "107000" @default.
- W2916237377 abstract "Genome-wide association and whole exome sequencing studies from Autism Spectrum Disorder (ASD) patient populations have implicated numerous risk factor genes whose mutation or deletion results in significantly increased incidence of ASD. Behavioral studies of monogenic mutant mouse models of ASD-associated genes have been useful for identifying aberrant neural circuitry. However, behavioral results often differ from lab to lab, and studies incorporating both males and females are often not performed despite the significant sex-bias of ASD. In this study, we sought to investigate the simple, passive behavior of home-cage activity monitoring across multiple 24-h days in four different monogenic mouse models of ASD: Shank3b-/-, Cntnap2-/-, Pcdh10+/-, and Fmr1 knockout mice. Relative to sex-matched wildtype (WT) littermates, we discovered significant home-cage hypoactivity, particularly in the dark (active) phase of the light/dark cycle, in male mice of all four ASD-associated transgenic models. For Cntnap2-/- and Pcdh10+/- mice, these activity alterations were sex-specific, as female mice did not exhibit home-cage activity differences relative to sex-matched WT controls. These home-cage hypoactivity alterations differ from activity findings previously reported using short-term activity measurements in a novel open field. Despite circadian problems reported in human ASD patients, none of the mouse models studied had alterations in free-running circadian period. Together, these findings highlight a shared phenotype across several monogenic mouse models of ASD, outline the importance of methodology on behavioral interpretation, and in some genetic lines parallel the male-enhanced phenotypic presentation observed in human ASDs." @default.
- W2916237377 created "2019-03-02" @default.
- W2916237377 creator A5017550855 @default.
- W2916237377 creator A5068649331 @default.
- W2916237377 creator A5078606655 @default.
- W2916237377 creator A5082649767 @default.
- W2916237377 creator A5086519148 @default.
- W2916237377 date "2019-11-01" @default.
- W2916237377 modified "2023-10-17" @default.
- W2916237377 title "Home-cage hypoactivity in mouse genetic models of autism spectrum disorder" @default.
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