Altered brain-wide auditory networks in a zebrafish model of fragile X syndrome.

Animals Autistic Disorder / genetics Brain / physiopathology Disease Models, Animal Fragile X Syndrome / genetics Zebrafish
Auditory perception Autism spectrum disorder Brain/physiopathology CGaMP Calcium imaging Fragile X syndrome Graph theory Light-sheet microscopy Sensory systems Zebrafish

Journal

BMC biology
ISSN: 1741-7007
Titre abrégé: BMC Biol
Pays: England
ID NLM: 101190720

Informations de publication

Date de publication:
16 09 2020
Historique:
received: 15 07 2020
accepted: 26 08 2020
entrez: 17 9 2020
pubmed: 18 9 2020
medline: 5 6 2021
Statut: epublish

Résumé

Loss or disrupted expression of the FMR1 gene causes fragile X syndrome (FXS), the most common monogenetic form of autism in humans. Although disruptions in sensory processing are core traits of FXS and autism, the neural underpinnings of these phenotypes are poorly understood. Using calcium imaging to record from the entire brain at cellular resolution, we investigated neuronal responses to visual and auditory stimuli in larval zebrafish, using fmr1 mutants to model FXS. The purpose of this study was to model the alterations of sensory networks, brain-wide and at cellular resolution, that underlie the sensory aspects of FXS and autism. Combining functional analyses with the neurons' anatomical positions, we found that fmr1 We demonstrated that fmr1

Sections du résumé

BACKGROUND
Loss or disrupted expression of the FMR1 gene causes fragile X syndrome (FXS), the most common monogenetic form of autism in humans. Although disruptions in sensory processing are core traits of FXS and autism, the neural underpinnings of these phenotypes are poorly understood. Using calcium imaging to record from the entire brain at cellular resolution, we investigated neuronal responses to visual and auditory stimuli in larval zebrafish, using fmr1 mutants to model FXS. The purpose of this study was to model the alterations of sensory networks, brain-wide and at cellular resolution, that underlie the sensory aspects of FXS and autism.
RESULTS
Combining functional analyses with the neurons' anatomical positions, we found that fmr1
CONCLUSIONS
We demonstrated that fmr1

Identifiants

DOI: 10.1186/s12915-020-00857-6 PMID: 32938458 PMC: PMC7493858
pubmed: 32938458
doi: 10.1186/s12915-020-00857-6
pii: 10.1186/s12915-020-00857-6
pmc: PMC7493858
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

125

Subventions

Organisme : Simons Foundation
ID : 399432
Pays : International
Organisme : NINDS NIH HHS
ID : RF1 NS118406
Pays : United States
Organisme : Simons Foundation
ID : 625793
Pays : International
Organisme : National Health and Medical Research Council
ID : APP1066887
Pays : International
Organisme : Australian Research Council
ID : DP110103612
Pays : International
Organisme : Australian Research Council
ID : DP140102036
Pays : International

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Auteurs

Lena Constantin (L)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.

Rebecca E Poulsen (RE)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.

Leandro A Scholz (LA)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.

Itia A Favre-Bulle (IA)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.
School of Mathematics and Physics, The University of Queensland, Brisbane, 4072, Australia.

Michael A Taylor (MA)

Australian Institute for Bioengineering and Nanotechnology, The University of Queensland, Brisbane, QLD, 4072, Australia.

Biao Sun (B)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.

Geoffrey J Goodhill (GJ)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia.
School of Mathematics and Physics, The University of Queensland, Brisbane, 4072, Australia.

Gilles C Vanwalleghem (GC)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia. g.vanwalleghem@uq.edu.au.

Ethan K Scott (EK)

Queensland Brain Institute, The University of Queensland, St Lucia, Brisbane, QLD, 4072, Australia. ethan.scott@uq.edu.au.
ORCID: 0000-0003-3150-9216

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Classifications MeSH

Recherchemedicale.com › Eucaryotes › Animaux › Altered brain-wide auditory networks in a...
Recherchemedicale.com › Troubles mentaux › Troubles du développement neurologique › Troubles généralisés du développement de l'enfant › Trouble du spectre autistique › Trouble autistique › Altered brain-wide auditory networks in a...
Recherchemedicale.com › Système nerveux › Système nerveux central › Encéphale › Altered brain-wide auditory networks in a...
Recherchemedicale.com › Techniques d'investigation › Modèles théoriques › Modèles biologiques › Modèles animaux de maladie humaine › Altered brain-wide auditory networks in a...
Recherchemedicale.com › Malformations et maladies congénitales, héréditaires et néonatales › Maladies génétiques congénitales › Maladies neurodégénératives héréditaires › Retard mental lié à l'X › Syndrome du chromosome X fragile › Altered brain-wide auditory networks in a...
Recherchemedicale.com › Eucaryotes › Animaux › Chordés › Vertébrés › Poissons › Cypriniformes › Cyprinidae › Danio zébré › Altered brain-wide auditory networks in a...