WARS1

Chr 14ARAD

tryptophanyl-tRNA synthetase 1

Also known as: GAMMA-2, HMN9, HMND9, IFI53, IFP53, NEDMSBA, WARS

The protein catalyzes the aminoacylation of tRNA with tryptophan in the cytoplasm, a critical step in protein synthesis. Mutations cause neurodevelopmental disorder with microcephaly and speech delay with or without brain abnormalities (autosomal recessive) and distal hereditary motor neuronopathy type 9 (autosomal dominant). The pathogenic mechanism involves disruption of cytoplasmic protein synthesis due to impaired tryptophan incorporation into nascent proteins.

OMIMResearchSummary from RefSeq, OMIM, UniProt
DNmechanismAR/ADLOEUF 0.492 OMIM phenotypes
Clinical SummaryWARS1
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Gene-Disease Validity (ClinGen)
distal hereditary motor neuropathy · ADLimited

Limited evidence — not for standalone diagnostic reporting

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.23) despite low pLI — interpret in context.
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ClinVar Variants
40 unique Pathogenic / Likely Pathogenic· 84 VUS of 186 total submissions
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.49LOEUF
pLI 0.303
Z-score 3.28
OE 0.23 (0.120.49)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.99Z-score
OE missense 0.67 (0.590.75)
188 obs / 282.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.23 (0.120.49)
00.351.4
Missense OE0.67 (0.590.75)
00.61.4
Synonymous OE0.88
01.21.6
LoF obs/exp: 5 / 21.4Missense obs/exp: 188 / 282.2Syn Z: 1.01
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveWARS1-related distal hereditary motor neuropathyOTHERAD
limitedWARS1-related neurodevelopmental syndromeOTHERAR
DN
0.6258th %ile
GOF
0.5759th %ile
LOF
0.3843th %ile

The highest-scoring mechanism for this gene is dominant-negative.

DNprediction above median · 1 literature citation

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.

Literature Evidence

DNCell transfection studies demonstrated a dominant-negative effect of the p.His257Arg mutation on aminoacylation activity of TrpRS, which subsequently compromised protein synthesis and reduced cell viability.PMID:28369220

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

ClinVar Variant Classifications

186 submitted variants in ClinVar

Classification Summary

Pathogenic36
Likely Pathogenic4
VUS84
Likely Benign24
Benign15
Conflicting5
36
Pathogenic
4
Likely Pathogenic
84
VUS
24
Likely Benign
15
Benign
5
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
0
1
35
0
36
Likely Pathogenic
1
2
1
0
4
VUS
3
74
7
0
84
Likely Benign
0
7
5
12
24
Benign
0
1
13
1
15
Conflicting
5
Total4856113168

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

View in ClinVar →

Protein Context — Lollipop Plot

WARS1 · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
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