GABRD

Chr 1AD

gamma-aminobutyric acid type A receptor subunit delta

Also known as: EIG10, EJM7, GABAARdelta, GEFSP5

The protein encodes the delta subunit of GABA-A receptors, which are pentameric ligand-gated chloride channels that mediate inhibitory neurotransmission in the brain. Mutations cause autosomal dominant generalized epilepsy with febrile seizures plus, type 5 through a predicted gain-of-function mechanism. The gene is highly intolerant to loss-of-function variants, consistent with the gain-of-function pathogenic mechanism.

OMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
GOFmechanismADLOEUF 0.241 OMIM phenotype
Clinical SummaryGABRD
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ADModerate

Moderate evidence — consider for supplementary testing

2 total gene-disease associations curated

Population Constraint (gnomAD)
Highly constrained gene — heterozygous loss-of-function variants are very rare in the population (pLI 0.99). One damaged copy is likely sufficient to cause disease.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

LoF intolerant — likely haploinsufficient
LoF Constraint
0.24LOEUF
pLI 0.993
Z-score 3.87
OE 0.05 (0.020.24)
Highly constrained

Highly LoF-intolerant (top ~10% of genes)

Missense Constraint
2.17Z-score
OE missense 0.64 (0.570.73)
189 obs / 293.5 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.05 (0.020.24)
00.351.4
Missense OE0.64 (0.570.73)
00.61.4
Synonymous OE1.24
01.21.6
LoF obs/exp: 1 / 19.4Missense obs/exp: 189 / 293.5Syn Z: -2.23
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
moderateGABRD-related neurodevelopmental disorder with epilepsyGOFAD
DN
0.6452th %ile
GOF
0.73top 25%
LOF
0.4726th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-of-function, loss-of-function and dominant-negative). Both the Badonyi & Marsh prediction and the broader genomic evidence point to gain-of-function as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

GOFprediction above median · 1 literature citation
LOFLOEUF 0.24
DNprediction 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.

Literature Evidence

GOFGain-of-function variants in GABRD reveal a novel pathway for neurodevelopmental disorders and epilepsy.PMID:34633442

Predictions from Badonyi M, Marsh JA. PLoS ONE. 2024;19(8):e0307312. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

GABRD · 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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