PUBLICATION

Ciliogenesis defects after neurulation impact brain development and neuronal activity in larval zebrafish

Authors
D'Gama, P.P., Jeong, I., Nygård, A.M., Trinh, A.T., Yaksi, E., Jurisch-Yaksi, N.
ID
ZDB-PUB-240613-8
Date
2024
Source
iScience   27: 110078110078 (Journal)
Registered Authors
Yaksi, Emre
Keywords
Biological sciences, Developmental neuroscience, Neuroscience
Datasets
GEO:GSE244171
MeSH Terms
none
PubMed
38868197 Full text @ iScience
Abstract
Cilia are slender, hair-like structures extending from cell surfaces and playing essential roles in diverse physiological processes. Within the nervous system, primary cilia contribute to signaling and sensory perception, while motile cilia facilitate cerebrospinal fluid flow. Here, we investigated the impact of ciliary loss on neural circuit development using a zebrafish line displaying ciliogenesis defects. We found that cilia defects after neurulation affect neurogenesis and brain morphology, especially in the cerebellum, and lead to altered gene expression profiles. Using whole brain calcium imaging, we measured reduced light-evoked and spontaneous neuronal activity in all brain regions. By shedding light on the intricate role of cilia in neural circuit formation and function in the zebrafish, our work highlights their evolutionary conserved role in the brain and sets the stage for future analysis of ciliopathy models.
Genes / Markers
Figures
Figure Gallery (7 images)
Show all Figures
Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
jf7TgTransgenic Insertion
    nns9TgTransgenic Insertion
      tp49d
        Point Mutation
        1 - 3 of 3
        Show
        Human Disease / Model
        Human Disease Fish Conditions Evidence
        ciliopathyTAS
        1 - 1 of 1
        Show
        Sequence Targeting Reagents
        No data available
        Fish
        Antibodies
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        DsRedEFGDsRed
        GCaMPEFGGCaMP
        1 - 2 of 2
        Show
        Mapping
        No data available