OXR1

Chr 8AR

oxidation resistance 1

Also known as: CHEGDD, Nbla00307, TLDC3

The OXR1 protein is an oxidoreductase that functions in the cellular response to oxidative stress and is located in mitochondria. Mutations cause autosomal recessive cerebellar hypoplasia/atrophy, epilepsy, and global developmental delay. This gene is highly constrained against loss-of-function variants (pLI 0.84, LOEUF 0.34), suggesting that functional copies are critical for normal development.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

Cerebellar hypoplasia/atrophy, epilepsy, and global developmental delayMIM #213000
AR
0
Active trials
15
Pubs (1 yr)
47
P/LP submissions
0%
P/LP missense
0.34
LOEUF· LoF intol.
LOF
Mechanism· G2P
Clinical SummaryOXR1
Population Constraint (gnomAD)
Moderately constrained gene (pLI 0.84) — some intolerance to loss-of-function variants.
📋
ClinVar Variants
44 unique Pathogenic / Likely Pathogenic· 121 VUS of 224 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.34LOEUF
pLI 0.841
Z-score 5.03
OE 0.20 (0.120.34)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
1.02Z-score
OE missense 0.86 (0.790.94)
380 obs / 440.0 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.20 (0.120.34)
00.351.4
Missense OE0.86 (0.790.94)
00.61.4
Synonymous OE1.09
01.21.6
LoF obs/exp: 9 / 45.7Missense obs/exp: 380 / 440.0Syn Z: -0.84

ClinVar Variant Classifications

224 submitted variants in ClinVar

Classification Summary

Pathogenic41
Likely Pathogenic3
VUS121
Likely Benign20
Benign3
Conflicting3
41
Pathogenic
3
Likely Pathogenic
121
VUS
20
Likely Benign
3
Benign
3
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
40
0
41
Likely Pathogenic
3
0
0
0
3
VUS
2
109
10
0
121
Likely Benign
1
5
2
12
20
Benign
0
0
1
2
3
Conflicting
3
Total71145314191

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

View in ClinVar →

Protein Context — Lollipop Plot

OXR1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold

Clinical Trials

Active and recruiting trials from ClinicalTrials.gov

No active trials found for this gene.

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Clinical Literature
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