AARS2

Chr 6AR

alanyl-tRNA synthetase 2, mitochondrial

Also known as: AARSL, COXPD8, LKENP, MT-ALARS, MTALARS

The encoded protein is a mitochondrial aminoacyl-tRNA synthetase that charges tRNAs with alanine during mitochondrial protein synthesis. Mutations cause combined oxidative phosphorylation deficiency 8 and progressive leukoencephalopathy with ovarian failure through autosomal recessive inheritance. The pathogenic mechanism involves impaired mitochondrial translation leading to defective oxidative phosphorylation.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismARLOEUF 0.832 OMIM phenotypes
Clinical SummaryAARS2
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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.
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GeneReview available — AARS2
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.83LOEUF
pLI 0.000
Z-score 2.56
OE 0.63 (0.480.83)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.07Z-score
OE missense 0.99 (0.931.06)
567 obs / 571.9 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.63 (0.480.83)
00.351.4
Missense OE0.99 (0.931.06)
00.61.4
Synonymous OE0.84
01.21.6
LoF obs/exp: 35 / 55.6Missense obs/exp: 567 / 571.9Syn Z: 1.88
DN
0.6841th %ile
GOF
0.6443th %ile
LOF
0.2679th %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

AARS2 · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗