PUBLICATION

Epb41l5 interacts with IQCB1 and regulates ciliary function in zebrafish embryos

Authors
Yu, T., Matsuda, M.
ID
ZDB-PUB-200606-28
Date
2020
Source
Journal of Cell Science   133(12): (Journal)
Registered Authors
Keywords
Cilia, Epb41l5, Epithelial morphogenesis, IQCB1, Left-right patterning, Zebrafish
MeSH Terms
  • Animals
  • Centrosome
  • Cilia*
  • Cytoskeletal Proteins
  • Zebrafish*/genetics
  • Zebrafish Proteins/genetics
(all 6)
PubMed
32501287 Full text @ J. Cell Sci.
Abstract
Erythrocyte protein band 4.1 like 5 (Epb41l5) is an adaptor protein beneath the plasma membrane that functions to control epithelial morphogenesis. Here we report a previously uncharacterized role of Epb41l5 in controlling ciliary function. We found that Epb41l5 forms a complex with IQCB1/NPHP5, a ciliopathy gene. Epb415 overexpression reduced IQCB1 localization at the ciliary base in cultured epithelial cells. Conversely, epb41l5 knockdown increased IQCB1 localization at the ciliary base. epb41l5-deficient zebrafish embryos or embryos expressing the C-terminally modified forms of Epb41l5 developed cilia with reduced motility and exhibited left-right patterning defects, an outcome of abnormal ciliary function. We observed genetic synergy between epb41l5 and iqcb1. Finally, Epb41l5 decreased IQCB1 interaction with Cep290, another ciliopathy gene and a component of the ciliary base and the centrosome. Together, these observations suggest that Epb41l5 regulates the composition of the ciliary base and the centrosome through IQCB1 and Cep290.
Genes / Markers
Marker Marker Type Name
dand5GENEDAN domain family, member 5
epb41l5GENEerythrocyte membrane protein band 4.1 like 5
hs6st3bGENEheparan sulfate 6-O-sulfotransferase 3b
iqcb1GENEIQ motif containing B1
myl7GENEmyosin, light chain 7, regulatory
ramp1GENEreceptor activity modifying protein 1
spawGENEsouthpaw
tp53GENEtumor protein p53
1 - 8 of 8
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Figures
No images available
Expression
No data available
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
b476
    		Deficiency
    hsc5TgTransgenic Insertion
      pt21TgTransgenic Insertion
        1 - 3 of 3
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        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Fish
        No data available
        Antibodies
        Orthology
        Engineered Foreign Genes
        Marker Marker Type Name
        GFPEFGGFP
        1 - 1 of 1
        Show
        Mapping
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        Citation
        Yu, T., Matsuda, M. (2020) Epb41l5 interacts with IQCB1 and regulates ciliary function in zebrafish embryos. Journal of Cell Science. 133(12):.