CACNB4

Chr 2AD

calcium voltage-gated channel auxiliary subunit beta 4

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

This gene encodes a member of the beta subunit family of voltage-dependent calcium channel complex proteins. Calcium channels mediate the influx of calcium ions into the cell upon membrane polarization and consist of a complex of alpha-1, alpha-2/delta, beta, and gamma subunits in a 1:1:1:1 ratio. Various versions of each of these subunits exist, either expressed from similar genes or the result of alternative splicing. The protein encoded by this locus plays an important role in calcium channel function by modulating G protein inhibition, increasing peak calcium current, controlling the alpha-1 subunit membrane targeting and shifting the voltage dependence of activation and inactivation. Certain mutations in this gene have been associated with idiopathic generalized epilepsy (IGE), juvenile myoclonic epilepsy (JME), and episodic ataxia, type 5. [provided by RefSeq, Aug 2016]

OMIMResearchGenerating clinical summary…
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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ClinVar Variants
5 unique Pathogenic / Likely Pathogenic· 189 VUS of 325 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available

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 OE?0.28 (0.170.49)
00.351.4
Missense OE?0.55 (0.490.63)
00.61.4
Synonymous OE?0.89
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

This gene — mechanism propensity

DN
0.74top 25%
GOF
0.6443th %ile
LOF
0.4726th %ile

This gene has evidence for multiple mechanisms of pathogenicity (dominant-negative, gain-of-function and loss-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
LOF20% of P/LP variants are LoF · LOEUF 0.49

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. Mechanism ranking also informed by gnomAD constraint, ClinVar, and ClinGen data.

ClinVar Variant Classifications

325 submitted variants in ClinVar

Classification Summary

Pathogenic3
Likely Pathogenic2
VUS189
Likely Benign61
Benign51
Conflicting17
3
Pathogenic
2
Likely Pathogenic
189
VUS
61
Likely Benign
51
Benign
17
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
1
0
2
0
3
Likely Pathogenic
0
2
0
0
2
VUS
3
105
78
3
189
Likely Benign
1
3
27
30
61
Benign
0
2
44
5
51
Conflicting
17
Total511215138323

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

View in ClinVar →

15 pathogenic / likely-pathogenic (of 21) ClinVar copy-number / structural variants overlap CACNB4 — these span large chromosomal regions, not the gene specifically, and are excluded from the counts above. Explore in CNV tools →

Protein Context — Lollipop Plot

CACNB4 · protein map & ClinVar variants

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