HCN2

Chr 19AD

hyperpolarization activated cyclic nucleotide gated potassium and sodium channel 2

Also known as: BCNG-2, BCNG2, EIG17, FEB2, GEFSP11, HAC-1

The protein is a hyperpolarization-activated cation channel that generates pacemaker currents in the heart and brain by conducting sodium and potassium ions. Mutations cause autosomal dominant epilepsy syndromes including generalized epilepsy with febrile seizures plus, familial febrile seizures, and idiopathic generalized epilepsy. Disease can result from multiple mechanisms depending on the specific variant, with some mutations potentially causing gain-of-function effects that alter normal channel properties.

Summary from RefSeq, OMIM, UniProt, Mechanism
Research Assistant →

Primary Disease Associations & Inheritance

{Epilepsy, idiopathic generalized, susceptibility to, 17}MIM #602477
AD
Febrile seizures, familial, 2MIM #602477
AD
Generalized epilepsy with febrile seizures plus, type 11MIM #602477
AD
0
Active trials
36
Pubs (1 yr)
44
P/LP submissions
29%
P/LP missense
0.45
LOEUF
Multiple*
Mechanism· predicted
Clinical SummaryHCN2
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Gene-Disease Validity (ClinGen)
complex neurodevelopmental disorder · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

3 total gene-disease associations curated

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.21) despite low pLI — interpret in context.
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ClinVar Variants
38 unique Pathogenic / Likely Pathogenic· 256 VUS of 394 total submissions

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Missense constrained — critical functional residues
LoF Constraint
0.45LOEUF
pLI 0.489
Z-score 3.53
OE 0.21 (0.110.45)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
3.41Z-score
OE missense 0.53 (0.470.59)
222 obs / 418.4 exp
Constrained

Highly missense-constrained (top ~0.1%)

Observed / Expected Ratios
LoF OE0.21 (0.110.45)
00.351.4
Missense OE0.53 (0.470.59)
00.61.4
Synonymous OE1.55
01.21.6
LoF obs/exp: 5 / 23.5Missense obs/exp: 222 / 418.4Syn Z: -5.75
DN
0.6841th %ile
GOF
0.83top 10%
LOF
0.48top 25%

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

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 HCN2 variants in genetic epilepsyPMID:29064616

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

ClinVar Variant Classifications

394 submitted variants in ClinVar

Classification Summary

Pathogenic32
Likely Pathogenic6
VUS256
Likely Benign56
Benign25
Conflicting7
32
Pathogenic
6
Likely Pathogenic
256
VUS
56
Likely Benign
25
Benign
7
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
10
21
0
32
Likely Pathogenic
0
1
5
0
6
VUS
8
233
15
0
256
Likely Benign
0
19
13
24
56
Benign
0
1
9
15
25
Conflicting
7
Total92646339382

LoF = frameshift, stop gained/lost, canonical splice · Counts from ClinVar esearch · Updated hourly

View in ClinVar →

Protein Context — Lollipop Plot

HCN2 · 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.

Search ClinicalTrials.gov →
Clinical Literature
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