RANBP1

Cilia effects upon perturbation of RANBP1

Cilia number / % ciliated:

Incrased cilia number

Loss-of-function effect:

Longer cilia

Overexpression effect:

Decreased

Ciliogenesis screen results (3 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.82) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-1.64) PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Mouse phenotype:

abnormal midbrain morphology, preweaning lethality, complete penetrance, abnormal embryo size, abnormal placenta size, abnormal hindbrain morphology, abnormal forebrain morphology, edema, abnormal craniofacial morphology

Mouse ciliopathy phenotype:

abnormal neural tube closure

Subcellular localization

basal body, centrosome, nucleus

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
• Actin & cytoskeleton regulation
• T cell biology
• Viral interactions

Function

Negative regulator of Ran GTP, as it stimulates Ran GTP to GDP conversion. RanBP1 Knockdown results in an increased concentration of Ran GTP at basal bodies, leading to ciliogenesis. Overexpression antagonizes primary cilia formation.