Fuzzy Homolog (FUZ)

Similar to knockdown of Xint (INTU), knockdown of Xfy disrupted Hedgehog signaling and caused defects in neural tube closure, including exencephaly, spina bifida, and holoprosencephaly-like features. The authors found that the Hedgehog defect in Xfy- and Xint-deficient embryos was secondary to a failure of ciliogenesis and that Xint and Xfy controlled the assembly of an apical actin network that is essential for the normal orientation of ciliary microtubules. Fluorescence microscopy revealed colocalization of Xint and Dishevelled at the apical surface of ciliated Xenopus epidermal cells. Park et al. (2006) concluded that Inturned, Fuzzy, and Dishevelled regulate ciliogenesis in Hedgehog-responsive cells through a conserved planar cell polarity (PCP) signaling pathway.