Abstract
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.
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Acknowledgements
The authors thank J. Reiter and R. Harland for critical discussions during this work. We also thank J. Hayes, S. Peyrot, A. Ellis for technical assistance; J. Sisson and J. Gross for critical reading; T. Holleman for the xVax1 plasmid; H. El-Hodiri for the xZic3 plasmid. J.B.W. dedicates this paper to J. Cruz. This work is supported by awards from the National Institute of General Medical Sciences/US National Institutes of Health and the Burroughs Wellcome Fund.
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Supplementary information
Supplementary Fig. 1
Sequence and expression pattern of Xint. (PDF 1450 kb)
Supplementary Fig. 2
Additional Xint morphant phenotypes. (PDF 2199 kb)
Supplementary Fig. 3
Sequence and expression pattern of Xenopus fuzzy. (PDF 1991 kb)
Supplementary Fig. 4
Quantification of convergent extennsion in Xint and Xfy morphants. (PDF 71 kb)
Supplementary Fig. 5
Xfy controls cytoskeletal organization during ciliogenesis. (PDF 890 kb)
Supplementary Table 1
Sequences of primer and morpholino-oligonucleotides used in this study. (PDF 23 kb)
Supplementary Video 1
Ciliated epidermal cell in control embryo. (MOV 409 kb)
Supplementary Video 2
Ciliated epidermal cell in Xint morphant. (MOV 321 kb)
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Park, T., Haigo, S. & Wallingford, J. Ciliogenesis defects in embryos lacking inturned or fuzzy function are associated with failure of planar cell polarity and Hedgehog signaling. Nat Genet 38, 303–311 (2006). https://doi.org/10.1038/ng1753
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DOI: https://doi.org/10.1038/ng1753
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