Ciliogenesis defects in embryos lacking inturned or fuzzy function are associated with failure of planar cell polarity and Hedgehog signaling

Nat Genet. 2006 Mar;38(3):303-11. doi: 10.1038/ng1753. Epub 2006 Feb 19.


The vertebrate planar cell polarity (PCP) pathway has previously been found to control polarized cell behaviors rather than cell fate. We report here that disruption of Xenopus laevis orthologs of the Drosophila melanogaster PCP effectors inturned (in) or fuzzy (fy) affected not only PCP-dependent convergent extension but also elicited embryonic phenotypes consistent with defective Hedgehog signaling. These defects in Hedgehog signaling resulted from a broad requirement for Inturned and Fuzzy in ciliogenesis. We show that these proteins govern apical actin assembly and thus control the orientation, but not assembly, of ciliary microtubules. Finally, accumulation of Dishevelled and Inturned near the basal apparatus of cilia suggests that these proteins function in a common pathway with core PCP components to regulate ciliogenesis. Together, these data highlight the interrelationships between cell polarity, cellular morphogenesis, signal transduction and cell fate specification.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Polarity / genetics*
  • Cilia / genetics*
  • Drosophila Proteins / genetics*
  • Drosophila melanogaster / embryology
  • Embryo, Nonmammalian / physiology*
  • Embryonic Development / genetics*
  • Hedgehog Proteins
  • Microtubules / physiology
  • Molecular Sequence Data
  • Nervous System / embryology
  • Phenotype
  • Signal Transduction
  • Xenopus Proteins / genetics
  • Xenopus laevis / embryology


  • Drosophila Proteins
  • Hedgehog Proteins
  • Xenopus Proteins
  • hh protein, Drosophila

Associated data

  • GENBANK/DQ355992
  • GENBANK/DQ357248