CEP128

centrosomal protein 128

Tier: Gold Standard Ciliary Genes

Ensembl:
ENSG00000100629
UniProt:
Q6ZU80
OMIM:
620667
Synonyms:
C14ORF145, C14ORF61

Cilia effects upon perturbation of CEP128

Loss-of-function effect:
Longer cilia
Overexpression effect:
Shorter cilia

Ciliogenesis screen results (4 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Failler/Valderrama et al. 2021 (siRNA) [siRNA]: Negative Regulator (Ciliogenesis Candidate) PMID:33206585
  • Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-3.06) PMID:41160700

Phenotypes

Mouse phenotype:
decreased prepulse inhibition, improved glucose tolerance, no spontaneous movement, decreased locomotor activity, decreased startle reflex, decreased total body fat amount, decreased grip strength

Subcellular localization

basal body, centrosome

References: PMID:29514088

Functional category

  • Ciliary assembly/disassembly
  • Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
  • Actin & cytoskeleton regulation
  • Reproduction & sperm
  • Cell migration & adhesion

Function

It encodes a subdistal appendage protein that regulates TGF-β/bone morphogenetic protein (BMP) signaling and RAB11 trafficking at the primary cilium. It has no major effect on centrosome structure but leads to increased ciliation frequency (29514088).