PUBLICATION

Wnt-PLC-IP3-Connexin-Ca2+ axis maintains ependymal motile cilia in zebrafish spinal cord

Authors
Zhang, J., Chandrasekaran, G., Li, W., Kim, D.Y., Jeong, I.Y., Lee, S.H., Liang, T., Bae, J.Y., Choi, I., Kang, H., Maeng, J.S., Kim, M.K., Lee, T., Park, S.W., Kim, M.J., Kim, H.S., Ro, H., Bae, Y.C., Park, H.C., Choi, E.Y., Choi, S.Y.
ID
ZDB-PUB-200422-170
Date
2020
Source
Nature communications   11: 1860 (Journal)
Registered Authors
Choi, Seok-Yong, Park, Hae-Chul
Keywords
none
MeSH Terms
  • Gene Knockout Techniques
  • Zebrafish/embryology*
  • Signal Transduction/genetics
  • Gene Expression Regulation, Developmental
  • Mice
  • Gap Junctions
  • Connexin 43/genetics
  • Connexin 43/metabolism*
  • Spinal Cord/metabolism*
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
  • Animals
  • Cell Differentiation
  • Connexins/metabolism*
  • Male
  • Mice, Knockout
  • Wnt Signaling Pathway/genetics
  • Ependyma/metabolism*
  • Ependyma/pathology
  • Humans
  • Cilia/genetics
  • Cilia/metabolism*
(all 22)
PubMed
32312952 Full text @ Nat. Commun.
Abstract
Ependymal cells (ECs) are multiciliated neuroepithelial cells that line the ventricles of the brain and the central canal of the spinal cord (SC). How ependymal motile cilia are maintained remains largely unexplored. Here we show that zebrafish embryos deficient in Wnt signaling have defective motile cilia, yet harbor intact basal bodies. With respect to maintenance of ependymal motile cilia, plcδ3a is a target gene of Wnt signaling. Lack of Connexin43 (Cx43), especially its channel function, decreases motile cilia and intercellular Ca2+ wave (ICW) propagation. Genetic ablation of cx43 in zebrafish and mice diminished motile cilia. Finally, Cx43 is also expressed in ECs of the human SC. Taken together, our findings indicate that gap junction mediated ICWs play an important role in the maintenance of ependymal motile cilia, and suggest that the enhancement of functional gap junctions by pharmacological or genetic manipulations may be adopted to ameliorate motile ciliopathy.
Genes / Markers
Figures
Figure Gallery (6 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
ABchemical treatment by environment: 6-bromoindirubin-3'-oximeLong-pec
ABchemical treatment by environment: Carbenoxolone sodiumLong-pec
ABchemical treatment by environment: Carbenoxolone sodiumLong-pec
ABchemical treatment by environment: DAPTLong-pec
ABchemical treatment by environment: DAPTLong-pec
ABchemical treatment by environment: IWR-1-endoLong-pec
ABchemical treatment by environment: IWR-1-endoLong-pec
ABchemical treatment by environment: IWR-1-endoLong-pec
ABchemical treatment by environment: thapsigarginDay 4
ABchemical treatment by environment: thapsigarginDay 4
1 - 10 of 54
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
cms5TgTransgenic Insertion
    cms6
      		Small Deletion
      hsc5TgTransgenic Insertion
        hsc16TgTransgenic Insertion
          t4
            		Deficiency
            w32TgTransgenic Insertion
              1 - 6 of 6
              Show
              Human Disease / Model
              No data available
              Sequence Targeting Reagents
              Target Reagent Reagent Type
              fzd7bMO2-fzd7bMRPHLNO
              gja1bCRISPR1-gja1bCRISPR
              gja1bMO6-gja1bMRPHLNO
              plcd3aMO1-plcd3aMRPHLNO
              wnt4bMO3-wnt4bMRPHLNO
              wnt5bMO1-wnt5bMRPHLNO
              wnt11MO4-wnt11MRPHLNO
              1 - 7 of 7
              Show
              Fish
              Fish
              AB
              AB + MO1-wnt5b
              AB + MO2-fzd7b
              AB + MO3-wnt4b
              AB + MO3-wnt4b + MO4-wnt11
              AB + MO4-wnt11
              AB + MO6-gja1b
              cms5Tg
              cms5Tg + MO6-gja1b
              Df(Chr07)t4/t4
              1 - 10 of 17
              Show
              Antibodies
              Orthology
              No data available
              Engineered Foreign Genes
              Marker Marker Type Name
              EGFPEFGEGFP
              GCaMPEFGGCaMP
              GFPEFGGFP
              1 - 3 of 3
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              Mapping
              No data available
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              Citation
              Zhang, J., Chandrasekaran, G., Li, W., Kim, D.Y., Jeong, I.Y., Lee, S.H., Liang, T., Bae, J.Y., Choi, I., Kang, H., Maeng, J.S., Kim, M.K., Lee, T., Park, S.W., Kim, M.J., Kim, H.S., Ro, H., Bae, Y.C., Park, H.C., Choi, E.Y., Choi, S.Y. (2020) Wnt-PLC-IP3-Connexin-Ca2+ axis maintains ependymal motile cilia in zebrafish spinal cord. Nature communications. 11:1860.