KCNS3

Chr 2

potassium voltage-gated channel modifier subfamily S member 3

Also known as: KV9.3

KCNS3 encodes a regulatory subunit that modulates delayed rectifier potassium channels by forming heterotetrameric channels with KCNB1 and slowing down channel deactivation and inactivation kinetics. Mutations cause autosomal dominant early infantile epileptic encephalopathy with prominent developmental delay and intellectual disability. The gene shows low constraint to loss-of-function variation, suggesting tolerance to such variants in the general population.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.91
Clinical SummaryKCNS3
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
0.91LOEUF
pLI 0.001
Z-score 1.83
OE 0.48 (0.270.91)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
1.02Z-score
OE missense 0.83 (0.740.92)
234 obs / 282.3 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.48 (0.270.91)
00.351.4
Missense OE0.83 (0.740.92)
00.61.4
Synonymous OE1.08
01.21.6
LoF obs/exp: 7 / 14.5Missense obs/exp: 234 / 282.3Syn Z: -0.70
DN
0.79top 25%
GOF
0.88top 5%
LOF
0.2093th %ile

This gene has evidence for multiple mechanisms of pathogenicity (gain-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
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.

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

KCNS3 · 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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