CACNA1H

Chr 16AD

calcium voltage-gated channel subunit alpha1 H

Also known as: CACNA1HB, Cav3.2, ECA6, EIG6, HALD4

The protein forms the pore-forming alpha-1 subunit of voltage-dependent T-type calcium channels, controlling calcium ion influx into cells upon membrane depolarization. Mutations cause childhood absence epilepsy, idiopathic generalized epilepsy, and familial hyperaldosteronism type IV through an autosomal dominant inheritance pattern. The pathogenic mechanism involves gain-of-function effects that disrupt normal calcium channel activity.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
MultiplemechanismADLOEUF 0.503 OMIM phenotypes
Clinical SummaryCACNA1H
Population Constraint (gnomAD)
Low constraint (pLI 0.00) — loss-of-function variants are relatively tolerated in the population.
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Clinical Trials
2 active or recruiting trials — potential therapeutic options may be available
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GeneReview available — CACNA1H
Authoritative clinical overview · Recommended first read
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Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.50LOEUF
pLI 0.000
Z-score 5.35
OE 0.38 (0.280.50)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
-2.36Z-score
OE missense 1.17 (1.131.22)
1717 obs / 1462.9 exp
Tolerant

Tolerant to missense variation

Observed / Expected Ratios
LoF OE0.38 (0.280.50)
00.351.4
Missense OE1.17 (1.131.22)
00.61.4
Synonymous OE1.65
01.21.6
LoF obs/exp: 32 / 85.3Missense obs/exp: 1717 / 1462.9Syn Z: -13.03
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedCACNA1H-related epilepsy, childhood absence, susceptibility toOTHERAD
DN
0.78top 25%
GOF
0.87top 5%
LOF
0.2679th %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

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

GOFRecently, a new autosomal-dominant form of familial PA, FH-IV, associated with mutations in the CACNA1H gene, was described in patients with hypertension and PA before the age of 10years.PMID:27315758

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

ClinVar Variant Classifications

0 submitted variants in ClinVar

Protein Context — Lollipop Plot

CACNA1H · 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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Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
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