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Table 1 Summary of proteins associated with canonical/non-canonical Wnt signaling and cilia

From: Cilia, Wnt signaling, and the cytoskeleton

Canonical Wnt

Cilia or Wnt association

Reference

Wnt proteins

  

Inv (NPHP2)

Ciliary localization and physical interaction with Dvl

[65, 66]

Dvl

Docking and polarization of basal bodies, basal body localization, targeted for degradation by Inv

[47, 66]

Cilia-associated proteins

 

Bbs-associated

Hyperactive Wnt response in knockout cell lines

[68]

Kif3aa

Up regulation of cellular Wnt response in knockout cell lines and mutant mice

[68, 69]

Ift88a

Up regulation of cellular Wnt response in mutant mice

[69, 70]

Ift40

Increase in expression of canonical Wnt pathway genes in kidney of mutant mice

[91]

Ift20

Increase in nuclear beta-catenin and expression of Wnt target genes in kidney of mutant mice

[89]

Ofd1

Up regulation of cellular Wnt response in mutant mice

[69]

Chibby

Binds beta-catenin preventing nuclear entry negatively regulating Wnt signaling

[71–73]

Seahorse

Binds to Dvl, constrains Wnt signaling in zebrafish

[74]

SREBP1c

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

[75]

Ahi1/Jbn

Abrogated Wnt signaling in kidney and cerebellum of mutant mice, facilitates beta-catenin entry into nucleus

[19, 106, 143]

Non-canonical Wnt

 

PCP effectors

 

Inturned

Highly expressed in ciliated tissue, required for ciliogenesis. Actin assembly, Rho localization, docking of basal bodies

[100, 144–146, 47, 147]

Fuzzy

Required for axoneme elongation, predicted role in vesicular trafficking

[100, 144]

Fritz

Expressed in ciliated tissue, required for ciliogenesis, mutations identified in human ciliopathies

[101]

Core PCP proteins

  

Dubroya

Regulates ciliogenesis, Apical Actin assembly

[148]

Frizzled

Defective ciliogenesis at zebrafish kupffers Vesicle

[148]

Dvl

Regulates ciliogenesis, Actin assembly, Rho localization, docking of basal bodies, associated with human ciliopathy proteins TMEM216 and TMEM67

[47, 149]

Celsr2/Celsr3

Regulates ciliogenesis in multiciliated ependymal cells via basal body docking at apical plasma membrane

[150]

Prickle

Regulates cilium length in zebrafish

[151]

Vangl2a

Localizes to some cilia, xenopus basal body localization and ciliogenesis, zebrafish conflicting data

[82, 83, 96]

  1. aFurther described in the legend
  2. The primary cilium was first implicated in suppression of canonical, β-catenin-dependent Wnt signaling, while being required for non-canonical Wnt signaling (PCP). One of the first studies to make this suggestion implicated inversin (Inv), a basal body protein associated with cystic kidney disease, as a molecular switch between the two Wnt signaling pathways. It was suggested that this occurred via targeting of cytoplasmic Dishevelled (Dvl) for degradation at the basal body. Another cilia-associated protein, the kinesin-like protein Kif3a, is thought to restrain canonical Wnt signaling by restricting the CK1-dependent phosphorylation of Dvl, which results in an active beta-catenin destruction complex, limiting beta-catenin induced transcription. It is possible that defects in protein interactions at the basal body, rather than in intraflagellar transport (IFT), could be responsible for dysregulation of Wnt signaling in cilia mutants. Such a suggestion is consistent with the findings that both IFT-mutant mice and mutant zebrafish do not show disruption of canonical Wnt signaling. aAxin 2 (Wnt target gene) and transgenic Wnt reporter normal in ift88, ift72, and kif3a mouse embryos [51]. No Wnt phenotype in ift88 zebrafish [52]. Conflicting zebrafish vangl2 data could be due to varying animal mutants and analysis methods.
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Cilia

ISSN: 2046-2530

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