GSDME

Chr 7AD

gasdermin E

Also known as: DFNA5, ICERE-1

The protein encoded by GSDME is a precursor of a pore-forming protein that converts non-inflammatory apoptosis to pyroptosis when cleaved and activated. Mutations cause nonsyndromic hearing impairment with autosomal recessive inheritance. The gene shows very high constraint against loss-of-function variants and is expressed in the fetal cochlea.

OMIMResearchSummary from RefSeq, UniProt
LOFmechanismADLOEUF 1.131 OMIM phenotype
Clinical SummaryGSDME
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Gene-Disease Validity (ClinGen)
nonsyndromic genetic hearing loss · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

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
1.13LOEUF
pLI 0.000
Z-score 1.12
OE 0.74 (0.501.13)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
-0.84Z-score
OE missense 1.15 (1.041.26)
304 obs / 265.4 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.74 (0.501.13)
00.351.4
Missense OE1.15 (1.041.26)
00.61.4
Synonymous OE1.10
01.21.6
LoF obs/exp: 16 / 21.6Missense obs/exp: 304 / 265.4Syn Z: -0.87
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongGSDME-related deafnessLOFAD
DN
0.5869th %ile
GOF
0.5563th %ile
LOF
0.2970th %ile

The Badonyi & Marsh model scores dominant-negative highest, but genomic evidence most strongly supports gain-of-function as the primary mechanism.

GOF1 literature citation

Literature Evidence

GOFIn transfection studies using HEK293T cells, Van Laer et al. (2004) found that posttransfection cell death approximately doubled when cells were transfected with mutant DFNA5-GFP compared to wildtype DFNA5-GFP, with cell death being attributed to necrotic rather than apoptotic events. This informatiPMID:15173223

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

GSDME · 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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