CCDC181

Cilia effects upon perturbation of CCDC181

Cilia number / % ciliated:

Severe reduction/essentially none

Loss-of-function effect:

Shorter cilia or No Flagella

Ciliogenesis screen results (1 screens)

• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680

Phenotypes

Mouse phenotype:

decreased total reti thickness, abnormal reti outer nuclear layer morphology

Subcellular localization

cilia, nucleus

References: PMID:41410385, PMID:39245650

Functional category

• Ciliary assembly/disassembly
• Actin & cytoskeleton regulation
• Reproduction & sperm
• Cell migration & adhesion

Function

CCDC181 is a structural component of the axonemal structure of sperm flagella as well as motile cilia. It might be a component of the apparatus necessary for the generation of ciliary motility. C-terminal region interacts with of the axoneme and Hook1, acting as a linker protein for phosphoprotein phosphatase 1 (PP1) to tack the phosphatase to the axonemal structure of cilia and flagella. Pronostic factor for prostate cancer. Ccdc181−/− mice displayed malformed flagella, including coiled, short, bent, absent, and irregular forms (PMID: 39245650)