GAS2

Chr 11AR

growth arrest specific 2

Also known as: DFNB125, GAS-2

The GAS2 protein is required to maintain microtubule bundles in inner ear supporting cells, providing mechanical stiffness necessary for sound transmission through the cochlea. Mutations cause autosomal recessive nonsyndromic hearing loss, with this gene being relatively tolerant to loss-of-function variants based on population constraint metrics. A GeneReviews entry is available for additional clinical guidance.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismARLOEUF 0.811 OMIM phenotype
Clinical SummaryGAS2
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
0.81LOEUF
pLI 0.001
Z-score 2.15
OE 0.45 (0.260.81)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
0.44Z-score
OE missense 0.91 (0.791.03)
154 obs / 170.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.45 (0.260.81)
00.351.4
Missense OE0.91 (0.791.03)
00.61.4
Synonymous OE0.97
01.21.6
LoF obs/exp: 8 / 17.8Missense obs/exp: 154 / 170.2Syn Z: 0.16
DN
0.7035th %ile
GOF
0.6541th %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

GAS2 · 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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