DYRK2

dual specificity tyrosine phosphorylation regulated kinase 2

Tier: Gold Standard Ciliary Genes

Ensembl:
ENSG00000127334
UniProt:
Q92630
OMIM:
603496

Cilia effects upon perturbation of DYRK2

Loss-of-function effect:
Longer cilia
Overexpression effect:
no effect

Ciliogenesis screen results (4 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.92) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: Increased Signaling (Negative Regulator) PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
  • Pusapati et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, neg_rank=363, lfc=-3.47) PMID:30270045

Phenotypes

Mouse phenotype:
decreased large unstained cell number

Ciliopathy associations

  • Skeletal Ciliopathy

References: PMID:34582081, PMID:32758357, PMID:39001875, PMID:40777246

Subcellular localization

basal body, nucleus

References: PMID:34259627, PMID:28242748

Functional category

  • Ciliary assembly/disassembly

Function

CEP78 interacts with the EDD1-DYRK2-DDB1VPRBP E3 ubiquitin ligase complex, which is involved in CP110 ubiquitination and degradation, and identified a novel interaction between CEP78 and CEP350 that is weakened by the CEP78L150S mutation(34259627). Cep78 controls centrosome homeostasis by inhibiting EDD‐DYRK2‐DDB1Vpr BP (28242748)