SCN4A

Chr 17ARAD

sodium voltage-gated channel alpha subunit 4

Also known as: CMS16, CMYO22A, CMYP22A, HOKPP2, HYKPP, HYPP, NAC1A, Na(V)1.4

SCN4A encodes the alpha subunit of a voltage-gated sodium channel expressed in skeletal muscle that generates and propagates action potentials. Mutations cause a spectrum of muscle disorders including periodic paralyses (hyperkalemic and hypokalemic), myotonias (paramyotonia congenita, acetazolamide-responsive myotonia congenita), congenital myopathies, and congenital myasthenic syndrome through gain-of-function mechanisms. Inheritance is autosomal dominant for most phenotypes, with autosomal recessive inheritance reported for some congenital myopathy presentations.

GeneReviewsOMIMResearchSummary from RefSeq, OMIM, UniProt, Mechanism
GOFmechanismAR/ADLOEUF 0.387 OMIM phenotypes
Clinical SummarySCN4A
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Gene-Disease Validity (ClinGen)
SCN4A-related myopathy, autosomal recessive · ARDefinitive

Definitive — sufficient evidence for diagnostic panels

Population Constraint (gnomAD)
Constrained for loss-of-function variants (OE-LoF 0.26) despite low pLI — interpret in context.
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ClinVar Variants
13 unique Pathogenic / Likely Pathogenic· 175 VUS of 300 total submissions
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Clinical Trials
1 active or recruiting trial — potential therapeutic options may be available
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GeneReview available — SCN4A
Authoritative clinical overview · Recommended first read
Open GeneReview ↗

Population Genetics & Constraint

gnomAD v4 — loss-of-function & missense intolerance

Moderate LoF intolerance
LoF Constraint
0.38LOEUF
pLI 0.013
Z-score 5.91
OE 0.26 (0.180.38)
Moderately constrained

More LoF-intolerant than ~75% of genes

Missense Constraint
1.56Z-score
OE missense 0.87 (0.820.92)
977 obs / 1124.2 exp
Tolerant

Mild missense constraint

Observed / Expected Ratios
LoF OE0.26 (0.180.38)
00.351.4
Missense OE0.87 (0.820.92)
00.61.4
Synonymous OE0.99
01.21.6
LoF obs/exp: 19 / 73.7Missense obs/exp: 977 / 1124.2Syn Z: 0.11
Curated Mechanism (G2P)Gene2Phenotype (DDG2P) ↗
definitiveSCN4A-related paramyotonia congenita of von EulenburgOTHERAD
definitiveSCN4A-related hyperkalemic periodic paralysisGOFAD
definitiveSCN4A-related hypokalemic periodic paralysisOTHERAD
DN
0.82top 10%
GOF
0.88top 5%
LOF
0.1895th %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

GOFTogether, our results suggest that N1366S is a gain-of-function mutation of NaV1.4 at low temperature and the mutation may be responsible for the clinical symptoms of paramyotonia congenita in the affected family and constitute a basis for studies into its pathogenesis.PMID:28940424

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

ClinVar Variant Classifications

300 submitted variants in ClinVar

Classification Summary

Pathogenic7
Likely Pathogenic6
VUS175
Likely Benign102
Conflicting1
7
Pathogenic
6
Likely Pathogenic
175
VUS
102
Likely Benign
1
Conflicting

Curated Variants Distribution

Classified variants from ClinVar · 5 ACMG categories

ClassificationLoFMissense + InframeNon-codingSynonymousTotal
Pathogenic
7
0
0
0
7
Likely Pathogenic
4
2
0
0
6
VUS
3
166
5
1
175
Likely Benign
0
0
36
66
102
Benign
0
0
0
0
0
Conflicting
1
Total141684167291

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

View in ClinVar →

Protein Context — Lollipop Plot

SCN4A · protein map & ClinVar variants

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

3D Protein StructureAlphaFold
Clinical Literature
Open Research Assistant →
Full-Text Mentions
NLP-detected gene mentions in article bodies · via PubTator3
PubTator3
Top 5 full-text resultsSearch PubTator3 ↗
Key Publications
Landmark & review papers · by relevance
PubMed
Top 5 results · since 2015Search PubMed ↗