CACNB4

Chr 2AD

calcium voltage-gated channel auxiliary subunit beta 4

Also known as: CAB4, CACNLB4, EA5, EIG9, EJM, EJM4, EJM6

This protein functions as the beta subunit of voltage-dependent calcium channels, increasing peak calcium current, shifting voltage dependencies of activation and inactivation, and controlling alpha-1 subunit membrane targeting. Mutations cause episodic ataxia type 5 and increase susceptibility to idiopathic generalized epilepsy and juvenile myoclonic epilepsy with autosomal dominant inheritance. The gene is highly constrained against loss-of-function variants (LOEUF 0.49), suggesting that complete loss of function is likely incompatible with normal development.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt
LOFmechanismADLOEUF 0.493 OMIM phenotypes
Clinical SummaryCACNB4
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Gene-Disease Validity (ClinGen)
epilepsy · ADRefuted

Refuted — evidence has disproved this relationship

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.28) 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 — CACNB4
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.49LOEUF
pLI 0.031
Z-score 3.77
OE 0.28 (0.170.49)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
2.69Z-score
OE missense 0.55 (0.490.63)
161 obs / 290.2 exp
Mild constraint

Moderately missense-constrained (top ~2.5%)

Observed / Expected Ratios
LoF OE0.28 (0.170.49)
00.351.4
Missense OE0.55 (0.490.63)
00.61.4
Synonymous OE0.89
01.21.6
LoF obs/exp: 9 / 32.0Missense obs/exp: 161 / 290.2Syn Z: 0.86
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
limitedCACNB4-related juvenile myoclonic epilepsyLOFAD
DN
0.74top 25%
GOF
0.6443th %ile
LOF
0.4726th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative and gain-of-function). Both the Badonyi & Marsh prediction and the broader genomic evidence point to dominant-negative as the predominant mechanism. Different variants in this gene may act through different mechanisms — interpret in context of the specific variant.

DNprediction above median
GOFprediction 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

CACNB4 · 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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