VPS41

VPS41 subunit of HOPS complex

Ensembl:
ENSG00000006715
UniProt:
P49754
OMIM:
605485
Synonyms:
HVSP41

Cilia effects upon perturbation of VPS41

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

Ciliogenesis screen results (5 screens)

  • Kim2016: No effect
  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-5.87) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-3.27) PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: Hyper-ciliogenesis (robust z=3.01, ciliated=60.7%) PMID:26595381
  • Pusapati et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, neg_rank=100, lfc=-3.06) PMID:30270045

Phenotypes

Human ciliopathy phenotype:
spinocerebellar ataxia, autosomal recessive 29

Subcellular localization

basal body, cytosol, endosome

Functional category

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

Function

The HOPS complex is a RAB19 effector functioning in CC and ciliogenesis through a non-lysosomal pathway. VPS41 and VPS11 co-precipitate with the C-terminus of TBC1D4 and this binding is significantly decreased in RAB19 KO cells. RAB19 recruits the HOPS complex to the centrosome and mediates interaction with TBC1D4 to coordi te CC and ciliary membrane growth during primary ciliogenesis(33561422).