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  • Review
  • Open Access

Cilia, Wnt signaling, and the cytoskeleton


  • Received: 11 October 2011
  • Accepted: 2 May 2012
  • Published:




  • Cilia
  • basal body
  • Wnt signaling
  • PCP
  • β-catenin
  • microtubules
  • actin
  • cytoskeleton
  • kinesins
  • GTPases





The Cilium - Structure and Function

Figure 1
Figure 1







Ciliary Proteins - Beyond the Cilium



Figure 2
Figure 2







Cilia in Wnt Signaling - the Importance of the Basal Body



One of the first indications that cilia might be involved in Wnt signaling was the identification of two key PCP components, Inversin (Nephrocystin 2) and Dishevelled (Dvl), at the basal body (Table 1) [47, 65, 66]. Mutations in Inversin causes one of the ciliopathies NPHP as well as situs inversus [67]. This was followed by observations that knockdown of several BBS and other ciliary-associated genes (Kif3a, Ift88, Ofd1) resulted in hyperactive canonical Wnt responses [6870]. Mutations in other ciliary-associated proteins also suggested a role for cilia in restraining Wnt signaling (Chibby, Seahorse, SREBP1c) [7175]. These differences might be attributed to tissue- and time-dependent variables, but also to the varying localization and function of these proteins.
Table 1


Canonical Wnt

Cilia or Wnt association


Wnt proteins


Inv (NPHP2)

Ciliary localization and physical interaction with Dvl

[65, 66]



[47, 66]

Cilia-associated proteins



Hyperactive Wnt response in knockout cell lines




[68, 69]


Up regulation of cellular Wnt response in mutant mice

[69, 70]








Up regulation of cellular Wnt response in mutant mice



Binds beta-catenin preventing nuclear entry negatively regulating Wnt signaling



Binds to Dvl, constrains Wnt signaling in zebrafish



Over expression disrupts ciliogenesis and increases canonical Wnt signaling in Xenopus.




[19, 106, 143]

Non-canonical Wnt


PCP effectors




[100, 144146, 47, 147]


Required for axoneme elongation, predicted role in vesicular trafficking

[100, 144]




Core PCP proteins



Regulates ciliogenesis, Apical Actin assembly



Defective ciliogenesis at zebrafish kupffers Vesicle




[47, 149]





Regulates cilium length in zebrafish




[82, 83, 96]

aFurther described in the legend



Non-canonical Wnt (PCP) signaling

The PCP signaling pathway was initially described in Drosophila and includes polarized localization of core PCP proteins, which ultimately leads to the establishment of polarized cell morphology [76, 77]. In vertebrates, PCP signaling has been shown to be required for convergent extension [78], defects in which result in disrupted neural tube closures, as well as misorientation of hair cells in the mammalian cochlea [79] and hair follicles in the epidermis [80]. One of the early indications that ciliary proteins may be linked to PCP regulation was the observation of PCP defects in ciliary mutant cochlea, linking basal body polarization with PCP regulation [81, 82]. In recent years PCP signaling has also been found to be required for the orientation of basal bodies in several other tissues, including positioning of motile monocilia at the node, gastrocoel, and Kupffer's vesicle, which is involved in the establishment of left-right asymmetry in fish [47, 8386].







Canonical Wnt signaling

Figure 3
Figure 3











Factors that regulate both canonical and non-canonical Wnt signaling


Figure 4
Figure 4














adenomatosis polyposis


Casein kinase 1


Bardet-Biedl syndrome


glycogen synthase 3b


intraflagellar transport




pericentriolar material


planar cell polarity


Sonic hedgehog.




Authors’ Affiliations



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