VARS2

Chr 6AR

valyl-tRNA synthetase 2, mitochondrial

This gene encodes a mitochondrial aminoacyl-tRNA synthetase that catalyzes the attachment of valine to tRNA(Val) for mitochondrial protein synthesis. Mutations cause combined oxidative phosphorylation deficiency-20 and early-onset mitochondrial encephalopathies through an autosomal recessive inheritance pattern. The pathogenic mechanism involves gain-of-function effects that disrupt mitochondrial translation and oxidative phosphorylation.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
GOFmechanismARLOEUF 0.691 OMIM phenotype
Clinical SummaryVARS2
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Gene-Disease Validity (ClinGen)
mitochondrial disease · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
Some data sources returned errors (1)

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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.69LOEUF
pLI 0.000
Z-score 3.73
OE 0.53 (0.410.69)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
2.39Z-score
OE missense 0.73 (0.670.79)
451 obs / 618.4 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.53 (0.410.69)
00.351.4
Missense OE0.73 (0.670.79)
00.61.4
Synonymous OE0.84
01.21.6
LoF obs/exp: 39 / 73.6Missense obs/exp: 451 / 618.4Syn Z: 2.02
DN
0.6065th %ile
GOF
0.6345th %ile
LOF
0.3066th %ile

The highest-scoring mechanism for this gene is gain-of-function.

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

VARS2 · 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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