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Altered fronto-striatal functions in the Gdi1-null mouse model of X-linked intellectual disability
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More, Lorenzo, Künnecke, Basil, Yekhlef, Latefa, Bruns, Andreas, Marte, Antonella, Fedele, Ernesto, Bianchi, Veronica, Taverna, Stefano, Gatti, Silvia and D'Adamo, Patrizia (2017) Altered fronto-striatal functions in the Gdi1-null mouse model of X-linked intellectual disability. Neuroscience, 344 . pp. 346-359. doi:10.1016/j.neuroscience.2016.12.043
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Official URL: http://dx.doi.org/10.1016/j.neuroscience.2016.12.0...
Abstract
RAB-GDP dissociation inhibitor 1 (GDI1) loss-of-function mutations are responsible for a form of non-specific X-linked Intellectual Disability (XLID) where the only clinical feature is cognitive impairment. GDI1 patients are impaired in specific aspects of executive functions and conditioned response, which are controlled by fronto-striatal circuitries. Previous molecular and behavioral characterization of the Gdi1-null mouse revealed alterations in the total number/distribution of hippocampal and cortical synaptic vesicles as well as hippocampal short-term synaptic plasticity, and memory deficits. In this study, we employed cognitive protocols with high translational validity to human condition that target the functionality of cortico-striatal circuitry such as attention and stimulus selection ability with progressive degree of complexity. We previously showed that Gdi1-null mice are impaired in some hippocampus-dependent forms of associative learning assessed by aversive procedures. Here, using appetitive-conditioning procedures we further investigated associative learning deficits sustained by the fronto-striatal system.
| Item Type: | Journal Article | ||||||||
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| Subjects: | Q Science > QP Physiology R Medicine > RC Internal medicine > RC0321 Neuroscience. Biological psychiatry. Neuropsychiatry |
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| Divisions: | Faculty of Science > Life Sciences (2010- ) | ||||||||
| Library of Congress Subject Headings (LCSH): | Executive functions (Neuropsychology), Conditioned response, Synaptic vesicles, Learning disabilities | ||||||||
| Journal or Publication Title: | Neuroscience | ||||||||
| Publisher: | Elsevier | ||||||||
| ISSN: | 0306-4522 | ||||||||
| Official Date: | 6 March 2017 | ||||||||
| Dates: |
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| Volume: | 344 | ||||||||
| Page Range: | pp. 346-359 | ||||||||
| DOI: | 10.1016/j.neuroscience.2016.12.043 | ||||||||
| Status: | Peer Reviewed | ||||||||
| Publication Status: | Published | ||||||||
| Access rights to Published version: | Open Access | ||||||||
| Funder: | Fondation Téléthon, F. Hoffmann-La Roche & Co. (FHLRCo.) | ||||||||
| Grant number: | TCP04015 (Fondation Téléthon), RPF #138 (F. Hoffmann-La Roche & Co. (FHLRCo.)) | ||||||||
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