GPX4

Chr 19AR

glutathione peroxidase 4

Also known as: GPx-4, GSHPx-4, MCSP, PHGPx, SMDS, snGPx, snPHGPx

The protein encoded by this gene belongs to the glutathione peroxidase family, members of which catalyze the reduction of hydrogen peroxide, organic hydroperoxides and lipid hydroperoxides, and thereby protect cells against oxidative damage. Several isozymes of this gene family exist in vertebrates, which vary in cellular location and substrate specificity. This isozyme has a high preference for lipid hydroperoxides and protects cells against membrane lipid peroxidation and cell death. It is also required for normal sperm development; thus, it has been identified as a 'moonlighting' protein because of its ability to serve dual functions as a peroxidase, as well as a structural protein in mature spermatozoa. Mutations in this gene are associated with Sedaghatian type of spondylometaphyseal dysplasia (SMDS). This isozyme is also a selenoprotein, containing the rare amino acid selenocysteine (Sec) at its active site. Sec is encoded by the UGA codon, which normally signals translation termination. The 3' UTRs of selenoprotein mRNAs contain a conserved stem-loop structure, designated the Sec insertion sequence (SECIS) element, that is necessary for the recognition of UGA as a Sec codon, rather than as a stop signal. Transcript variants resulting from alternative splicing or use of alternate promoters have been described to encode isoforms with different subcellular localization. [provided by RefSeq, Dec 2018]

OMIMResearchGenerating clinical summary…
LOFmechanismARLOEUF 1.281 OMIM phenotype
Clinical SummaryGPX4
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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ClinVar Variants
17 unique Pathogenic / Likely Pathogenic· 92 VUS of 235 total submissions
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Clinical Trials
3 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint?
1.28LOEUF
pLI 0.000
Z-score 0.91
OE 0.71 (0.411.28)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint?
-1.08Z-score
OE missense 1.27 (1.121.45)
159 obs / 125.2 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios?
LoF OE?0.71 (0.411.28)
00.351.4
Missense OE?1.27 (1.121.45)
00.61.4
Synonymous OE?1.59
01.21.6
LoF obs/exp: 8 / 11.3Missense obs/exp: 159 / 125.2Syn Z: -3.47
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongGPX4-related spondylometaphyseal dysplasia, Sedaghatian typeLOFAR

This gene — mechanism propensity

Predictions shown for reference only — model trained on dominant genes, not applicable to AR conditions.

DN
0.76top 25%
GOF
0.6541th %ile
LOF
0.2679th %ile

The Badonyi & Marsh prediction model was trained exclusively on dominant disease genes. Predictions are not reliable for genes with autosomal recessive inheritance and are shown at reduced opacity for reference only.

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312.

ClinVar Variant Classifications

235 submitted variants in ClinVar

Classification Summary

Pathogenic8
Likely Pathogenic9
VUS92
Likely Benign83
Benign26
Conflicting4
8
Pathogenic
9
Likely Pathogenic
92
VUS
83
Likely Benign
26
Benign
4
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
6
0
2
0
8
Likely Pathogenic
8
1
0
0
9
VUS
1
86
4
1
92
Likely Benign
0
11
42
30
83
Benign
0
1
20
5
26
Conflicting
4
Total15996836222

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

32 pathogenic / likely-pathogenic (of 44) ClinVar copy-number / structural variants overlap GPX4 — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

GPX4 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.