Synaptic plasticity and spatial working memory are impaired in the CD mouse model of Williams-Beuren syndrome
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- dc.contributor.author Borralleras Fumaña, Cristina, 1988-ca
- dc.contributor.author Mato, Susanaca
- dc.contributor.author Amédée, Thierryca
- dc.contributor.author Matute, Carlosca
- dc.contributor.author Mulle, Christopheca
- dc.contributor.author Pérez Jurado, Luis Albertoca
- dc.contributor.author Campuzano Uceda, María Victoriaca
- dc.date.accessioned 2016-11-08T17:16:49Z
- dc.date.available 2016-11-08T17:16:49Z
- dc.date.issued 2016ca
- dc.description.abstract Mice heterozygous for a complete deletion (CD) equivalent to the most common deletion found in individuals with Williams-Beuren syndrome (WBS) recapitulate relevant features of the neurocognitive phenotype, such as hypersociability, along with some neuroanatomical alterations in specific brain areas. However, the pathophysiological mechanisms underlying these phenotypes still remain largely unknown. We have studied the synaptic function and cognition in CD mice using hippocampal slices and a behavioral test sensitive to hippocampal function. We have found that long-term potentiation (LTP) elicited by theta burst stimulation (TBS) was significantly impaired in hippocampal field CA1 of CD animals. This deficit might be associated with the observed alterations in spatial working memory. However, we did not detect changes in presynaptic function, LTP induction mechanisms or AMPA and NMDA receptor function. Reduced levels of Brain-derived neurotrophic factor (BDNF) were present in the CA1-CA3 hippocampal region of CD mice, which could account for LTP deficits in these mice. Taken together, these results suggest a defect of CA1 synapses in CD mice to sustain synaptic strength after stimulation. These data represent the first description of synaptic functional deficits in CD mice and further highlights the utility of the CD model to study the mechanisms underlying the WBS neurocognitive profile.
- dc.description.sponsorship This work was supported by the Spanish Ministry of Economy and Competitiveness (grant SAF2012-40036 to V.C. and SAF2013-45084-R to C.M.), CIBERNED (Grant PRY-15-404 to C.M.), “Programa de Excelencia Maria de Maeztu” MDM-2014-0370, the Generalitat de Catalunya (2014SGR1468 and ICREA Acadèmia to L.A.P.-J.).
- dc.format.mimetype application/pdfca
- dc.identifier.citation Borralleras C, Mato S, Amédée T, Matute C, Mulle C, Pérez-Jurado L, Campuzano V. Synaptic plasticity and spatial working memory are impaired in the CD mouse model of Williams-Beuren syndrome. Molecular Brain. 2016; 9(1):76. DOI: 10.1186/s13041-016-0258-7ca
- dc.identifier.doi http://dx.doi.org/10.1186/s13041-016-0258-7
- dc.identifier.issn 1756-6606ca
- dc.identifier.uri http://hdl.handle.net/10230/27469
- dc.language.iso engca
- dc.publisher BioMed Centralca
- dc.relation.ispartof Molecular Brain. 2016;9(1):76
- dc.relation.projectID info:eu-repo/grantAgreement/ES/3PN/SAF2012-40036
- dc.rights © 2016 The Author(s). Open Access. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.ca
- dc.rights.accessRights info:eu-repo/semantics/openAccessca
- dc.rights.uri http://creativecommons.org/licenses/by/4.0/
- dc.subject.keyword Williams-Beuren syndrome
- dc.subject.keyword Mouse model
- dc.subject.keyword Memory
- dc.subject.keyword Synaptic plasticity
- dc.subject.keyword LTP
- dc.subject.keyword Hippocampus
- dc.title Synaptic plasticity and spatial working memory are impaired in the CD mouse model of Williams-Beuren syndromeca
- dc.type info:eu-repo/semantics/articleca
- dc.type.version info:eu-repo/semantics/publishedVersionca