PCP and Wnt pathway components act in parallel during zebrafish mechanosensory hair cell orientation

Nat Commun. 2019 Sep 5;10(1):3993. doi: 10.1038/s41467-019-12005-y.


Planar cell polarity (PCP) plays crucial roles in developmental processes such as gastrulation, neural tube closure and hearing. Wnt pathway mutants are often classified as PCP mutants due to similarities between their phenotypes. Here, we show that in the zebrafish lateral line, disruptions of the PCP and Wnt pathways have differential effects on hair cell orientations. While mutations in the PCP genes vangl2 and scrib cause random orientations of hair cells, mutations in wnt11f1, gpc4 and fzd7a/b induce hair cells to adopt a concentric pattern. This concentric pattern is not caused by defects in PCP but is due to misaligned support cells. The molecular basis of the support cell defect is unknown but we demonstrate that the PCP and Wnt pathways work in parallel to establish proper hair cell orientation. Consequently, hair cell orientation defects are not solely explained by defects in PCP signaling, and some hair cell phenotypes warrant re-evaluation.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Cell Polarity / genetics*
  • Cell Polarity / physiology*
  • Gene Expression Regulation, Developmental
  • Hair Cells, Auditory / metabolism*
  • Heparan Sulfate Proteoglycans / genetics
  • Membrane Proteins / genetics
  • Morphogenesis / genetics
  • Morphogenesis / physiology
  • Mutation
  • Neural Tube Defects / genetics
  • Neurulation / genetics
  • Receptors, Cell Surface / genetics
  • Wnt Signaling Pathway / genetics*
  • Wnt Signaling Pathway / physiology*
  • Wnt1 Protein / genetics
  • Zebrafish / genetics*
  • Zebrafish Proteins / genetics


  • Heparan Sulfate Proteoglycans
  • Membrane Proteins
  • Receptors, Cell Surface
  • Wnt1 Protein
  • Zebrafish Proteins
  • fzd7a protein, zebrafish
  • fzd7b protein, zebrafish
  • gpc4 protein, zebrafish
  • scrib protein, zebrafish
  • vangl2 protein, zebrafish
  • wnt1 protein, zebrafish