Kirshenbaum, GS, Idris, NF, Dachtler, J et al. (2 more authors) (2016) Deficits in social behavioral tests in a mouse model of alternating hemiplegia of childhood. Journal of Neurogenetics, 30 (1). pp. 42-49. ISSN 0167-7063
Abstract
Social behavioral deficits have been observed in patients diagnosed with alternating hemiplegia of childhood (AHC), rapid-onset dystonia-parkinsonism and CAPOS syndrome, in which specific missense mutations in ATP1A3, encoding the Na+, K+-ATPase α3 subunit, have been identified. To test the hypothesis that social behavioral deficits represent part of the phenotype of Na+, K+-ATPase α3 mutations, we assessed the social behavior of the Myshkin mouse model of AHC, which has an I810N mutation identical to that found in an AHC patient with co-morbid autism. Myshkin mice displayed deficits in three tests of social behavior: nest building, pup retrieval and the three-chamber social approach test. Chronic treatment with the mood stabilizer lithium enhanced nest building in wild-type but not Myshkin mice. In light of previous studies revealing a broad profile of neurobehavioral deficits in the Myshkin model – consistent with the complex clinical profile of AHC – our results suggest that Na+, K+-ATPase α3 dysfunction has a deleterious, but nonspecific, effect on social behavior. By better defining the behavioral profile of Myshkin mice, we identify additional ATP1A3-related symptoms for which the Myshkin model could be used as a tool to advance understanding of the underlying neural mechanisms and develop novel therapeutic strategies.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © 2016 The Author(s). Published by Informa UK Limited, trading as Taylor & Francis Group. This is an Accepted Manuscript of an article published by Taylor & Francis in Journal of Neurogenetics on 8 June 2016, available online: http://www.tandfonline.com/10.1080/01677063.2016.1182525. Uploaded in accordance with the publisher's self-archiving policy. |
Keywords: | Social behavior; ATP1A3; Alternating hemiplegia of childhood; Mouse model |
Dates: |
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Institution: | The University of Leeds |
Academic Units: | The University of Leeds > Faculty of Biological Sciences (Leeds) > School of Biomedical Sciences (Leeds) |
Depositing User: | Symplectic Publications |
Date Deposited: | 20 Jun 2016 11:30 |
Last Modified: | 18 Jul 2017 11:37 |
Published Version: | http://dx.doi.org/10.1080/01677063.2016.1182525 |
Status: | Published |
Publisher: | Taylor & Francis |
Identification Number: | 10.1080/01677063.2016.1182525 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:100834 |