RAB23

RAB23, member RAS oncogene family

Ensembl:
ENSG00000112210
UniProt:
Q9ULC3
OMIM:
606144

Cilia effects upon perturbation of RAB23

Cilia number / % ciliated:
Decreased cilia number
Loss-of-function effect:
Shorter cilia
Overexpression effect:
Increased

Ciliogenesis screen results (4 screens)

  • Kim2016: Not Reported
  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-3.34) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-2.94) PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Mouse phenotype:
decreased bone mineral content, increased mean platelet volume, decreased lean body mass, preweaning lethality, complete penetrance
Mouse ciliopathy phenotype:
increased circulating alanine transamise level, increased circulating aspartate transamise level

Ciliopathy associations

  • Carpenter Syndrome

Subcellular localization

basal body, cilia

Functional category

  • Ciliary assembly/disassembly
  • Trafficking (BBSome, small GTPases, vesicular transport, ATPases)
  • Signaling (Hedgehog, GPCRs, ion channels)

Function

Negative regulator of the Hedgehog sig ling pathway, required for retrograde transport away from primary cilia (17646400). It mediates the ciliary recycle of Smo (20375059). Facilitates binding of cytiplasmatic Kif17 with ciliary import carrier importin 尾2, essential for its targeting to the primary cilium (26136363). Rab23-CKO mice exhibit cell-type specific ciliary abnormalities in chondrocytes, MEFs, neural progenitors. Rab23-KO NPCs show perturbed ciliation and desensitized Hh signaling. Loss causes shortened or absent cilia in context-dependent manner.

Model organism evidence

Mus musculus (5 references)

Rab23 GTPase and IFT43 regulate the trafficking of prostaglandin E receptor 4 to primary cilia.

RAB23 loss-of-function mutation causes context-dependent ciliopathy in Carpenter syndrome.

PMIDs: 41372612, 40825043, 39615683, 34210780, 29727300

Danio rerio (2 references)

Rab23 GTPase and IFT43 regulate the trafficking of prostaglandin E receptor 4 to primary cilia.

RAB23 loss-of-function mutation causes context-dependent ciliopathy in Carpenter syndrome.

PMIDs: 41372612, 40825043