CUL9

Cilia effects upon perturbation of CUL9

Ciliogenesis screen results (2 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-4.31) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680

Phenotypes

Mouse phenotype:

increased circulating potassium level; increased circulating lactate dehydrogenase level; increased blood urea nitrogen level; enlarged spleen; abnormal vertebral arch morphology; vertebral transformation; abnormal vertebrae morphology; abnormal spleen morphology; enlarged thymus; increased circulating creatine kinase level; abnormal thymus morphology

Mouse ciliopathy phenotype:

abnormal kidney morphology

Subcellular localization

cilia, cytosol

Function

The collection of regulators has diverse distributions throughout the cell with Bfar, Cop1, Cul9, Fancg, Skp1a, Uba3, Ube2l3, and Wwp1 showing weak localization to cilia. In addition, Sytl4 localized to the ciliary pocket at the base of the cilium, and Fbxw11 localized to centriolar satellites Positive regulator of Hedgehog signaling (PMID: 34161574).