MAK

male germ cell associated kinase

Ensembl:
ENSG00000111837
UniProt:
P20794
OMIM:
154235
Synonyms:
DJ417M14.2, RP62

Cilia effects upon perturbation of MAK

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

Ciliogenesis screen results (3 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Human ciliopathy phenotype:
retinitis pigmentosa; retinitis pigmentosa 62

Ciliopathy associations

  • Retinal Dystrophy/Degeneration

Subcellular localization

basal body, centrosome, cilia, nucleus

Functional category

  • Ciliary assembly/disassembly
  • Actin & cytoskeleton regulation
  • Signaling (Hedgehog, GPCRs, ion channels)
  • Transition zone

Function

Involved in the regulation of photoreceptor connecting cilium length. Mutations cause autosomal domi nt Retinitis Pigmentosa (22110072). Essential for the regulation of ciliary length, preventing excessive elongation of the cilia and is required for the long-term survival of photoreceptors (21148103). Forms a complex with DYF-5 to regulate cilia morphology ikely via modulation of microtubule stability (30955935).

Model organism evidence

C. elegans (2 references)

DYF-5 regulates intraflagellar transport by affecting train turnaround.

We identify several new mutations in genes encoding proteins functioning in intraflagellar transport and cilia assembly, in components of BBSome, MAPK-15, and DYF-5 kinases.

PMIDs: 40072497, 39854273

Mus musculus (2 references)

Ccrk-Mak/Ick signaling is a ciliary transport regulator essential for retinal photoreceptor survival.

Interestingly, zebrafish mak mutants failed to generate axonemes during photoreceptor ciliogenesis, whereas basal bodies were specified.

PMIDs: 39293864, 38813692

Danio rerio (1 reference)

Interestingly, zebrafish mak mutants failed to generate axonemes during photoreceptor ciliogenesis, whereas basal bodies were specified.

PMIDs: 38813692