FTO

FTO alpha-ketoglutarate dependent dioxygenase

Ensembl:
ENSG00000140718
UniProt:
Q9C0B1
OMIM:
610966
Synonyms:
ALKBH9, IFEX9, KIAA1752, MGC5149

Cilia effects upon perturbation of FTO

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

Ciliogenesis screen results (4 screens)

  • Kim2016: Not Reported
  • Wheway et al. 2015 (siRNA) [siRNA]: Hyper-ciliogenesis (z=2.04) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Mouse phenotype:
decreased circulating cholesterol level, decreased circulating iron level, decreased circulating alanine transamise level
Human ciliopathy phenotype:
obesity

Subcellular localization

cilia associated gene, nucleus

Functional category

  • Ciliary assembly/disassembly
  • T cell biology
  • ECM & connective tissue

Function

Fto knockout mice display aberrant tissue specific cilia. These data identify FTO as a protein-regulator of the balanced activation between canonical and non-canonical branches of the Wnt pathway ( 24503721). R demethylation by FTO stabilizes the FOXJ1 mR for proper motile ciliogenesis ( 33761320).

Model organism evidence

Xenopus (1 reference)

RNA demethylation by FTO stabilizes the FOXJ1 mRNA for proper motile ciliogenesis.

PMIDs: 33761320

Danio rerio (1 reference)

Loss of FTO antagonises Wnt signaling and leads to developmental defects associated with ciliopathies.

PMIDs: 24503721