SYNJ1

synaptojanin 1

Ensembl:
ENSG00000159082
UniProt:
O43426
OMIM:
604297
Synonyms:
INPP5G, PARK20

Cilia effects upon perturbation of SYNJ1

Cilia number / % ciliated:
Incrased cilia number
Loss-of-function effect:
Longer cilia

Ciliogenesis screen results (2 screens)

  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Mouse phenotype:
tremors, decreased red blood cell distribution width, decreased neutrophil cell number, increased lymphocyte cell number, decreased monocyte cell number, preweaning lethality, complete penetrance
Mouse ciliopathy phenotype:
persistence of hyaloid vascular system

Subcellular localization

basal body, cytosol, endosome

Functional category

  • Ciliary assembly/disassembly
  • Actin & cytoskeleton regulation

Function

SYNJ1, (Sy ptojanin-1 (SJ1)) may contribute to the control of ciliary protein dy mics in dopaminergic neurons, with implications on cilia-mediated sig ling (38635628).

Model organism evidence

Danio rerio (1 reference)

The outer segments and connecting cilia of nrc(a14) cone photoreceptors are normal, but RibeyeB and VAMP2/synaptobrevin, which normally localize to the synapse, accumulate in the nrc(a14) inner segment.

PMIDs: 24392132