GPX4

glutathione peroxidase 4

Ensembl:
ENSG00000167468
UniProt:
P36969
OMIM:
138322
Synonyms:
MCSP, PHGPX

Cilia effects upon perturbation of GPX4

Ciliogenesis screen results (4 screens)

  • Kim2016: Not Reported
  • 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

Mouse phenotype:
preweaning lethality, complete penetrance, embryonic lethality prior to tooth bud stage, embryonic lethality prior to organogenesis
Mouse ciliopathy phenotype:
hydrometra

Ciliopathy associations

  • Spondylometaphyseal Dysplasia

Subcellular localization

cilia associated gene, cytosol, mitochondria, nucleus

Functional category

  • Ciliary assembly/disassembly

Function

Mutations in the GPX4 gene cause Spondylometaphyseal Dysplasia (PMID: 9556300, PMID:24706940).

Model organism evidence

Mus musculus (2 references)

Photoreceptor cells before undergoing apoptosis (P11) exhibited decreased mitochondrial biomass, decreased number of connecting cilia, as well as disorganized structure of outer segments.

These impairments were accompanied by the structural abnormality, such as a hairpin-like flagella bend at the midpiece and swelling of mitochondria in the spermatozoa.

PMIDs: 22207760, 19783653