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PMID: 23536755 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

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

PloS one ·Vol. 8 ·No. 3 ·2013-00-00 ·Pages e51456

Ng MC, Yang YL, Lu KT

Abstract

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.

MeSH Terms
Animals Animals, Genetically Modified Anxiety Avoidance Learning Behavior, Animal Disease Models, Animal Fragile X Mental Retardation Protein/genetics,metabolism Fragile X Syndrome/genetics,metabolism,physiopathology Gene Knockout Techniques Genotype Humans Hyperkinesis/genetics Long-Term Potentiation Long-Term Synaptic Depression Phenotype Synapses/metabolism Synaptic Transmission/genetics
Chemicals
Fragile X Mental Retardation Protein
Authors & Affiliations
3 authors, click to expand affiliations / ORCID
Ng Ming-Chong
Department of Life Science, National Taiwan Normal University, Taipei, Taiwan.
Yang Yi-Ling
Lu Kwok-Tung
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Article Info
Journal
PloS one
Abbr.
PLoS One
ISSN
1932-6203
DOI
10.1371/journal.pone.0051456
Published
2013-00-00
Epub
2013-00-11
Pages
e51456
Language
English
Region
United States
NLM ID
101285081
PMCID
PMC3594205
Subset
IM
Grants
Wellcome Trust · United Kingdom
External Links
PubMed source DOI: 10.1371/journal.pone.0051456 PMC Full Text View journal details
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