KCNF1

Chr 2

potassium voltage-gated channel modifier subfamily F member 1

Also known as: IK8, KCNF, KV5.1, kH1

The protein functions as a regulatory alpha-subunit of voltage-gated potassium channels that modulates the expression and gating kinetics of KCNB1 and KCNB2 channels by affecting deactivation and inactivation processes. Mutations in KCNF1 cause early infantile epileptic encephalopathy, an autosomal recessive disorder characterized by seizure onset in the first months of life. The gene shows moderate constraint against loss-of-function variants (LOEUF 0.66), and the severe early-onset neurological phenotype reflects the critical role of potassium channel regulation in neuronal excitability.

OMIMResearchSummary from RefSeq, UniProt
MultiplemechanismLOEUF 0.66
Clinical SummaryKCNF1
Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.25) despite low pLI — interpret in context.

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Missense constrained — critical functional residues
LoF Constraint
0.66LOEUF
pLI 0.246
Z-score 2.37
OE 0.25 (0.120.66)
Tolerant

Typical tolerance to LoF variation

Missense Constraint
3.38Z-score
OE missense 0.48 (0.420.55)
160 obs / 333.8 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.25 (0.120.66)
00.351.4
Missense OE0.48 (0.420.55)
00.61.4
Synonymous OE0.90
01.21.6
LoF obs/exp: 3 / 11.8Missense obs/exp: 160 / 333.8Syn Z: 0.98
DN
0.75top 25%
GOF
0.86top 5%
LOF
0.2386th %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

KCNF1 · 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.

Search ClinicalTrials.gov →
Clinical Literature
Open Research Assistant →
Key Publications
Landmark & review papers · by relevance
PubMed
Kv5.1 antibody in epilepsy patients with unknown etiology.
Küçükali Cİ et al.·Epilepsy Res
2022Cohort
Top 1 results · since 2015Search PubMed ↗
Recent Gene-Specific Literature
Gene in title · MEDLINE · newest first
Europe PMC
KCNF1 promotes lung cancer by modulating ITGB4 expression.
Chen CY et al.·Cancer Gene Ther
2023Open Access