KCNE5

Chr X

potassium voltage-gated channel subfamily E regulatory subunit 5

Also known as: KCNE1L

The protein functions as an ancillary subunit that regulates voltage-gated potassium channels, particularly serving as an inhibitory beta-subunit of the cardiac potassium channel KCNQ1. Mutations cause AMME complex (a neurodevelopmental disorder with autism, microcephaly, mild intellectual disability, and epilepsy) and are associated with Brugada syndrome 6, a cardiac arrhythmia disorder. The gene shows tolerance to loss-of-function variants (LOEUF 1.83), suggesting variable penetrance or that some variants may act through other mechanisms.

Summary from RefSeq, OMIM, UniProt
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Primary Disease Associations & Inheritance

AMME complexMIM #300194
?Brugada syndrome 6MIM #613119
0
Active trials
3
Pubs (1 yr)
0
P/LP submissions
P/LP missense
1.83
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryKCNE5
🧬
Gene-Disease Validity (ClinGen)
Brugada syndrome · ADDisputed

Disputed — evidence questions this relationship

Population Constraint (gnomAD)
Low constraint (pLI 0.12) — loss-of-function variants are relatively tolerated in the population.
📖
GeneReview available — KCNE5
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Tolerant — LoF & missense variants common in population
LoF Constraint
1.83LOEUF
pLI 0.119
Z-score 0.40
OE 0.65 (0.201.83)
Tolerant

Highly tolerant — LoF variants common in population

Missense Constraint
0.41Z-score
OE missense 0.86 (0.701.07)
60 obs / 69.5 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.65 (0.201.83)
00.351.4
Missense OE0.86 (0.701.07)
00.61.4
Synonymous OE0.72
01.21.6
LoF obs/exp: 1 / 1.5Missense obs/exp: 60 / 69.5Syn Z: 1.32
DN
0.6455th %ile
GOF
0.83top 10%
LOF
0.3648th %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

KCNE5 · protein map & ClinVar variants

Showing all ClinVar variants across the protein. Search a specific variant to highlight its position.

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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗