KCTD10

Cilia effects upon perturbation of KCTD10

Cilia number / % ciliated:

Decreased cilia number

Loss-of-function effect:

Impaired ciliogenesis

Ciliogenesis screen results (5 screens)

• Kim2016: Not Reported
• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-2.23) PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, neg_rank=74, lfc=-2.71) PMID:30270045

Phenotypes

Mouse phenotype:

abnormal radius morphology, decreased circulating sodium level, decreased circulating chloride level, decreased grip strength, trunk curl, preweaning lethality, complete penetrance, abnormal joint morphology

Subcellular localization

basal body, nucleus

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)

Function

Component of cullin-3鈥. Through several screening steps, we identified the cullin-3–RBX1–KCTD10 complex as the E3 ligase that mediates CEP97 degradation and removal from the mother centriole. Depletion of each component of this E3 complex caused aberrant accumulation of CEP97 on the centrosome, suppressed the removal of CEP97 and CP110 from the mother centriole, and impaired ciliogenesis. KCTD10 was specifically localized to the mother centriole, and it was detected as a single dot adjacent to γ-tubulin at the base of the primary cilium in serum-starved cells. (30404837)