GLI2

Cilia effects upon perturbation of GLI2

Loss-of-function effect:

Longer cilia

Ciliogenesis screen results (6 screens)

• Kim2016: Not Reported
• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-8.90) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-2.85) PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, neg_rank=4, lfc=-4.38) PMID:30270045
• Kim et al. 2010 (siRNA) [siRNA]: Shorter Cilia (Area per Cilia z=-2.54) PMID:20393562

Phenotypes

Human ciliopathy phenotype:

postaxial polydactyly-anterior pituitary anomalies-facial dysmorphism syndrome

Ciliopathy associations

• Holoprosencephaly

Subcellular localization

basal body, cilia, nucleus

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Actin & cytoskeleton regulation
• T cell biology
• Neurogenesis & migration
• Cell migration & adhesion
• Signaling (Hedgehog, GPCRs, ion channels)
• Axon guidance & growth
• Transcription regulation
• Cilia length regulation

Function

Hedgehog sig ling pathway effector. Cilia or interflagellar transport proteins are required for GLI2 function and regulation (21209912). Regulates autophagy- mediated Ofd1 removal to control primary cilia formation and length (30463852). GLI2 knockdown/knockout leads to increased cilia length in NIH3T3 fibroblasts after serum starvation. GLI2 also participates in ciliary trafficking for Hedgehog signaling. (30782777,40073114)