OVCH1

Chr 12

ovochymase 1

The OVCH1 protein functions as an ovochymase, a serine protease involved in protein processing. Mutations cause autosomal recessive primary ovarian insufficiency, leading to infertility and hypogonadism in females. This gene shows low constraint against loss-of-function variants, consistent with its recessive inheritance pattern and tissue-specific expression primarily affecting reproductive function.

0
Active trials
1
Pubs (1 yr)
0
P/LP submissions
P/LP missense
1.22
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryOVCH1
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
Some data sources returned errors (1)

ncbi: SyntaxError: Bad control character in string literal in JSON at position 94 (line 1 column 95)

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.22LOEUF
pLI 0.000
Z-score 0.16
OE 0.98 (0.791.22)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.38Z-score
OE missense 0.95 (0.891.02)
543 obs / 568.7 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.98 (0.791.22)
00.351.4
Missense OE0.95 (0.891.02)
00.61.4
Synonymous OE0.98
01.21.6
LoF obs/exp: 55 / 56.3Missense obs/exp: 543 / 568.7Syn Z: 0.18
DN
0.6648th %ile
GOF
0.6444th %ile
LOF
0.4136th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction above median

Note: In-silico variant effect predictors (SIFT, PolyPhen, REVEL, CADD) may underestimate pathogenicity of missense variants in genes with GOF or DN mechanisms. Consider functional evidence and clinical context.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

OVCH1 · protein map & ClinVar variants

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

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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Key Publications
Landmark & review papers · by relevance
PubMed
Top 3 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC

No open access results found