CRB2

Cilia effects upon perturbation of CRB2

Ciliogenesis screen results (5 screens)

• Kim2016: Not Reported
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
• Pusapati et al. 2018 (CRISPR) [CRISPR]: Negative Regulator (Hh signaling, pos_rank=240, lfc=0.88) PMID:30270045
• Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-4.54) PMID:41160700

Phenotypes

Mouse phenotype:

preweaning lethality; complete penetrance; abnormal vibrissa morphology

Mouse ciliopathy phenotype:

abnormal retina morphology

Human ciliopathy phenotype:

ventriculomegaly-cystic kidney disease

Subcellular localization

cilia associated gene

Functional category

• Metabolism; Motile cilium & axoneme; Trafficking (BBSome, small GTPases, vesicular transport, ATPases); Ciliary assembly/disassembly

Function

CRB2 depletion blocks the formation of the ciliary vesicle after activation of the mother centriole, indicating a defect in either transport or anchoring of the ciliary vesicle. CRB2 is accumulated in intracellular vesicles in the vicinity of the centrosome and we showed here that CRB2 forms a complex made of HOOK2 and PAR6α, two proteins important for primary ciliogenesis or centrosome integrity. In addition, CRB2 interacts with RAB8A which over-expression rescued CRB2 knockdown. All together, these findings indicate that CRB2 regulates a membrane transport step essential for the formation of the ciliary vesicle.

Model organism evidence