KCNC3

Chr 19AD

potassium voltage-gated channel subfamily C member 3

Also known as: KSHIIID, KV3.3, SCA13

The protein forms a voltage-gated potassium channel that mediates rapid repolarization of fast-firing brain neurons, particularly regulating action potential frequency, shape and duration in Purkinje cells. Mutations cause spinocerebellar ataxia 13 through an autosomal dominant inheritance pattern. The pathogenic mechanism involves gain-of-function mutations that disrupt normal neuronal excitability and cerebellar function.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismADLOEUF 0.591 OMIM phenotype
Clinical SummaryKCNC3
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Gene-Disease Validity (ClinGen)
spinocerebellar ataxia type 13 · ADDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.26) despite low pLI — interpret in context.
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
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GeneReview available — KCNC3
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.59LOEUF
pLI 0.206
Z-score 2.72
OE 0.26 (0.130.59)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.04Z-score
OE missense 0.57 (0.510.64)
224 obs / 393.8 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.26 (0.130.59)
00.351.4
Missense OE0.57 (0.510.64)
00.61.4
Synonymous OE1.03
01.21.6
LoF obs/exp: 4 / 15.6Missense obs/exp: 224 / 393.8Syn Z: -0.28
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
strongKCNC3-related spinocerebellar ataxiaOTHERAD
DN
0.7033th %ile
GOF
0.84top 5%
LOF
0.4332th %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 · 1 literature citation
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

DNKCNC3(R420H), located in the voltage sensor of the channel, has no detectable channel activity when expressed alone, and strong dominant negative effects when coexpressed with wild-type KCNC3PMID:18592334
GOFGain-of function or dominant-negative mutations in the voltage-gated potassium channel KCNC3 (Kv3.3) were recently identified as a cause of autosomal dominant spinocerebellar ataxia.PMID:21479265

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

KCNC3 · 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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