Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome

Abstract Fragile X syndrome (FXS) is an X-chromosome linked intellectual disability and the most common known inherited single gene cause of autism spectrum disorder (ASD). Building upon demonstrated deficits in neuronal plasticity and spatial memory in FXS, we investigated how spatial information p...

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Autores principales:	Tara Arbab, Cyriel M. A. Pennartz, Francesco P. Battaglia
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2018
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/ce5093c598a146a28babb7dacf706418
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spelling	oai:doaj.org-article:ce5093c598a146a28babb7dacf7064182021-12-02T15:08:13ZImpaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome10.1038/s41598-018-26853-z2045-2322https://doaj.org/article/ce5093c598a146a28babb7dacf7064182018-06-01T00:00:00Zhttps://doi.org/10.1038/s41598-018-26853-zhttps://doaj.org/toc/2045-2322Abstract Fragile X syndrome (FXS) is an X-chromosome linked intellectual disability and the most common known inherited single gene cause of autism spectrum disorder (ASD). Building upon demonstrated deficits in neuronal plasticity and spatial memory in FXS, we investigated how spatial information processing is affected in vivo in an FXS mouse model (Fmr1-KO). Healthy hippocampal neurons (so-called place cells) exhibit place-related activity during spatial exploration, and their firing fields tend to remain stable over time. In contrast, we find impaired stability and reduced specificity of Fmr1-KO spatial representations. This is a potential biomarker for the cognitive dysfunction observed in FXS, informative on the ability to integrate sensory information into an abstract representation and successfully retain this conceptual memory. Our results provide key insight into the biological mechanisms underlying cognitive disabilities in FXS and ASD, paving the way for a targeted approach to remedy these.Tara ArbabCyriel M. A. PennartzFrancesco P. BattagliaNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 8, Iss 1, Pp 1-9 (2018)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Tara Arbab Cyriel M. A. Pennartz Francesco P. Battaglia Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
description	Abstract Fragile X syndrome (FXS) is an X-chromosome linked intellectual disability and the most common known inherited single gene cause of autism spectrum disorder (ASD). Building upon demonstrated deficits in neuronal plasticity and spatial memory in FXS, we investigated how spatial information processing is affected in vivo in an FXS mouse model (Fmr1-KO). Healthy hippocampal neurons (so-called place cells) exhibit place-related activity during spatial exploration, and their firing fields tend to remain stable over time. In contrast, we find impaired stability and reduced specificity of Fmr1-KO spatial representations. This is a potential biomarker for the cognitive dysfunction observed in FXS, informative on the ability to integrate sensory information into an abstract representation and successfully retain this conceptual memory. Our results provide key insight into the biological mechanisms underlying cognitive disabilities in FXS and ASD, paving the way for a targeted approach to remedy these.
format	article
author	Tara Arbab Cyriel M. A. Pennartz Francesco P. Battaglia
author_facet	Tara Arbab Cyriel M. A. Pennartz Francesco P. Battaglia
author_sort	Tara Arbab
title	Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
title_short	Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
title_full	Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
title_fullStr	Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
title_full_unstemmed	Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome
title_sort	impaired hippocampal representation of place in the fmr1-knockout mouse model of fragile x syndrome
publisher	Nature Portfolio
publishDate	2018
url	https://doaj.org/article/ce5093c598a146a28babb7dacf706418
work_keys_str_mv	AT taraarbab impairedhippocampalrepresentationofplaceinthefmr1knockoutmousemodeloffragilexsyndrome AT cyrielmapennartz impairedhippocampalrepresentationofplaceinthefmr1knockoutmousemodeloffragilexsyndrome AT francescopbattaglia impairedhippocampalrepresentationofplaceinthefmr1knockoutmousemodeloffragilexsyndrome
_version_	1718388214145744896

Impaired hippocampal representation of place in the Fmr1-knockout mouse model of fragile X syndrome

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