SCN5A Variant R376C

Summary of observed carriers, functional annotations, and structural context for SCN5A R376C. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

2%

0/11 effective observations

Estimated BrS1 penetrance

24%

2/11 effective observations

Total carriers

1

0 BrS1 · 0 LQT3 · 1 unaffected

R376C has not been reported in gnomAD. This residue resides in a Mild_Hotspot region for Brugada syndrome and a Non_Hotspot region for LQT3.

Variant features alone are equivalent to phenotyping 2 individuals for Brugada syndrome and 0 individuals for LQT3.

In silico predictors

Variant-level computational predictors.
PROVEAN PolyPhen-2 BLAST-PSSM REVEL Penetrance Density BrS (%) Penetrance Density LQT3 (%)
-5 1 -3.78 0.941 29 2

PROVEAN scores below -2 suggest deleterious impact. REVEL scores above 0.5–0.75 are often interpreted as likely pathogenic. PolyPhen-2 scores above 0.85 are typically pathogenic. Penetrance density summarises neighbouring residue risk (Kroncke et al. 2019).

Reported carrier data

Observed carriers by publication or cohort.
Source Year Carriers Unaffected LQT3 BrS1 Other Other Disease
20129283 2010 1 0 0 0
29325976 2018 1 0 1 0
30059973 2018 3 3 0 0
Literature, cohort, and gnomAD 1 1 0 0
Variant features alone 15 13 0 2

Totals may differ from individual publications due to duplicate patients removed during curation.

Functional data

Peak and late/persistent current values are relative to wild-type (100% indicates no change). V1/2 activation and inactivation denote the membrane potentials (mV) at which half-maximal current is achieved.

Published electrophysiology measurements.
PubMed ID Year Cell Type Peak Current (% WT) V1/2 Activation (mV) V1/2 Inactivation (mV) Late/Persistent Current (% WT)
20129283 2010
29325976 2018
30059973 2018

Nearby variants

Neighbouring residues within 15 Ångström provide structural context. Variants listed in the right-most column have been observed clinically or in gnomAD.

Previously observed variants near R376C.
Neighbour residue Distance (Å) Observed variants
364 12
1702 14
901 10 E901K, S901L,
276 11 L276Q, L276P,
363 14
348 8 P348A,
279 15
355 14 F355I, F355C,
1715 12
1687 10
372 10
371 13 Q371E,
1711 11 c.5131delG,
904 11 W904X,
1706 11 Q1706H, Q1706H,
1716 13 p.L1716SfsX71
376 0 R376C, R376H,
1714 14 D1714G,
384 13 S384T,
1688 14
354 12
897 15 G897R, G897R, G897E,
1423 15 D1423H,
1692 12
1422 15 M1422R,
378 9
902 14
349 7 D349N,
373 7
1712 10 G1712S, G1712C,
379 6
1703 14
898 12
893 15 R893C, R893H,
1709 14 p.T1709del, T1709R, T1709M,
325 11 L325R,
1420 13 G1420R, G1420D, G1420V, G1420P,
900 10
324 10
393 13
1713 15
383 11
323 13
347 12
382 12
351 12 G351S, G351C, G351D, G351V,
374 9 W374G,
1689 12 D1689N,
350 11 H350Q, H350Q,
903 14 p.M903CfsX29,
367 9 R367C, R367H, R367L,
370 15 T370M,
381 10 c.1140+1G>A, c.1141-3C>A,
1686 12
322 13
905 14
375 5
1691 14
352 14 Y352C,
368 12
899 13
1710 14 S1710L,
380 6
377 6
1419 12 K1419E,
353 9 T353I,