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- W2777394414 abstract "Mutant mouse models of neurodevelopmental disorders with intellectual disabilities provide useful translational research tools, especially in cases where robust cognitive deficits are reproducibly detected. However, motor, sensory and/or health issues consequent to the mutation may introduce artifacts that preclude testing in some standard cognitive assays. Touchscreen learning and memory tasks in small operant chambers have the potential to circumvent these confounds. Here we use touchscreen visual discrimination learning to evaluate performance in the maternally derived Ube3a mouse model of Angelman syndrome, the Ts65Dn trisomy mouse model of Down syndrome, and the Mecp2Bird mouse model of Rett syndrome. Significant deficits in acquisition of a 2-choice visual discrimination task were detected in both Ube3a and Ts65Dn mice. Procedural control measures showed no genotype differences during pretraining phases or during acquisition. Mecp2 males did not survive long enough for touchscreen training, consistent with previous reports. Most Mecp2 females failed on pretraining criteria. Significant impairments on Morris water maze spatial learning were detected in both Ube3a and Ts65Dn, replicating previous findings. Abnormalities on rotarod in Ube3a, and on open field in Ts65Dn, replicating previous findings, may have contributed to the observed acquisition deficits and swim speed abnormalities during water maze performance. In contrast, these motor phenotypes do not appear to have affected touchscreen procedural abilities during pretraining or visual discrimination training. Our findings of slower touchscreen learning in 2 mouse models of neurodevelopmental disorders with intellectual disabilities indicate that operant tasks offer promising outcome measures for the preclinical discovery of effective pharmacological therapeutics." @default.
- W2777394414 created "2018-01-05" @default.
- W2777394414 creator A5063501456 @default.
- W2777394414 creator A5069126678 @default.
- W2777394414 date "2018-02-15" @default.
- W2777394414 modified "2023-10-14" @default.
- W2777394414 title "Touchscreen learning deficits in<i>Ube3a</i>, Ts65Dn and<i>Mecp2</i>mouse models of neurodevelopmental disorders with intellectual disabilities" @default.
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- W2777394414 doi "https://doi.org/10.1111/gbb.12452" @default.
- W2777394414 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/6013336" @default.
- W2777394414 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/29266714" @default.
- W2777394414 hasPublicationYear "2018" @default.
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