Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.

Fragile X syndrome (FXS) is the most frequent inherited form of human mental retardation. It is characterized by cognitive impairment and physical and behavioral problems and is caused by the silencing of fmr1 transcription and the absence of the fmr1 protein (FMRP). Recently, animal models of FXS h...

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Autores principales:	Ming-Chong Ng, Yi-Ling Yang, Kwok-Tung Lu
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2013
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/384b2bcdb69b4dd994589536d501b71b
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spelling	oai:doaj.org-article:384b2bcdb69b4dd994589536d501b71b2021-11-18T07:54:02ZBehavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.1932-620310.1371/journal.pone.0051456https://doaj.org/article/384b2bcdb69b4dd994589536d501b71b2013-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/23536755/?tool=EBIhttps://doaj.org/toc/1932-6203Fragile X syndrome (FXS) is the most frequent inherited form of human mental retardation. It is characterized by cognitive impairment and physical and behavioral problems and is caused by the silencing of fmr1 transcription and the absence of the fmr1 protein (FMRP). Recently, animal models of FXS have greatly facilitated the investigation of the molecular and cellular mechanisms of this loss-of-function disorder. The present study was aimed to further characterize the role of FMRP in behavior and synaptic function by using fmr1 knockout zebrafish. In adult zebrafish, we found that fmr1 knockout produces the anxiolytic-like responses of increased exploratory behavior in light/dark and open-field tests and avoidance learning impairment. Furthermore, electrophysiological recordings from telencephalic slice preparations of knockout fish displayed markedly reduced long-term potentiation and enhanced long-term depression compared to wild-type fish; however, basal glutamatergic transmission and presynaptic function at the lateral (Dl) and medial (Dm) division of the dorsal telencephalon synapse remained normal. Taken together, our study not only evaluates the mechanism of FRMP but also suggests that zebrafish have valuable potential as a complementary vertebrate model in studying the molecular pathogenesis of human fragile X syndrome.Ming-Chong NgYi-Ling YangKwok-Tung LuPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 8, Iss 3, p e51456 (2013)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Ming-Chong Ng Yi-Ling Yang Kwok-Tung Lu Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
description	Fragile X syndrome (FXS) is the most frequent inherited form of human mental retardation. It is characterized by cognitive impairment and physical and behavioral problems and is caused by the silencing of fmr1 transcription and the absence of the fmr1 protein (FMRP). Recently, animal models of FXS have greatly facilitated the investigation of the molecular and cellular mechanisms of this loss-of-function disorder. The present study was aimed to further characterize the role of FMRP in behavior and synaptic function by using fmr1 knockout zebrafish. In adult zebrafish, we found that fmr1 knockout produces the anxiolytic-like responses of increased exploratory behavior in light/dark and open-field tests and avoidance learning impairment. Furthermore, electrophysiological recordings from telencephalic slice preparations of knockout fish displayed markedly reduced long-term potentiation and enhanced long-term depression compared to wild-type fish; however, basal glutamatergic transmission and presynaptic function at the lateral (Dl) and medial (Dm) division of the dorsal telencephalon synapse remained normal. Taken together, our study not only evaluates the mechanism of FRMP but also suggests that zebrafish have valuable potential as a complementary vertebrate model in studying the molecular pathogenesis of human fragile X syndrome.
format	article
author	Ming-Chong Ng Yi-Ling Yang Kwok-Tung Lu
author_facet	Ming-Chong Ng Yi-Ling Yang Kwok-Tung Lu
author_sort	Ming-Chong Ng
title	Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
title_short	Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
title_full	Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
title_fullStr	Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
title_full_unstemmed	Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.
title_sort	behavioral and synaptic circuit features in a zebrafish model of fragile x syndrome.
publisher	Public Library of Science (PLoS)
publishDate	2013
url	https://doaj.org/article/384b2bcdb69b4dd994589536d501b71b
work_keys_str_mv	AT mingchongng behavioralandsynapticcircuitfeaturesinazebrafishmodeloffragilexsyndrome AT yilingyang behavioralandsynapticcircuitfeaturesinazebrafishmodeloffragilexsyndrome AT kwoktunglu behavioralandsynapticcircuitfeaturesinazebrafishmodeloffragilexsyndrome
_version_	1718422778309246976

Behavioral and synaptic circuit features in a zebrafish model of fragile X syndrome.

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