SCN5A Variant S1458Y

Summary of observed carriers, functional annotations, and structural context for SCN5A S1458Y. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

58%

3/11 effective observations

Estimated BrS1 penetrance

20%

2/11 effective observations

Total carriers

1

0 BrS1 · 1 LQT3 · 0 unaffected

S1458Y has not been reported in gnomAD. This residue resides in a Mild_Hotspot region for Brugada syndrome and a Hotspot region for LQT3.

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

In silico predictors

Variant-level computational predictors.
PROVEAN PolyPhen-2 BLAST-PSSM REVEL Penetrance Density BrS (%) Penetrance Density LQT3 (%)
-5.24 0.782 -3.6 0.964 24 70

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
15840476 2005 1 1 0 0
25904541 2015 1 1 0 0
Literature, cohort, and gnomAD 1 0 1 0
Variant features alone 15 11 2 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)
15840476 2005
25904541 2015

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 S1458Y.
Neighbour residue Distance (Å) Observed variants
896 8 C896S, C896S,
937 15
895 11 L895F,
1417 7
1765 13
839 14 L839P,
842 14
1340 13 V1340I,
894 14 I894M,
1457 4
1453 7
1455 7
1757 15
1449 13 Y1449S, Y1449C,
1452 11
1756 14 I1756V,
1461 6 T1461S, T1461S,
926 14
1344 12 F1344L, F1344S, F1344L, F1344L,
1450 11
1411 13
1451 10 V1451L, V1451D,
934 10
1458 0 S1458Y,
933 15
935 11 L935P,
897 12 G897R, G897R, G897E,
1348 12 F1348L, F1348L, F1348L,
1464 10 c.4389_4396delCCTCTTTA, L1464P,
927 12 N927S, N927K, N927K,
1466 12 c.4396_4397insG,
1422 14 M1422R,
1418 7
892 11 F892I,
1341 10
898 13
893 14 R893C, R893H,
1462 5
938 12
1412 10 L1412F,
1420 14 G1420R, G1420D, G1420V, G1420P,
843 13 T843A,
1456 8
930 13 c.2787+17_2787+18insACACACACACACACACACACACA, c.2788-6C>T,
1459 4 c.4376_4379delTCTT,
1460 8 F1460L, F1460L, F1460L,
1425 14
1713 14
1454 5
1338 14 L1338V,
1448 15 I1448L, I1448T,
1409 14 Y1409C, Y1409X,
1421 11
1345 9 W1345C, W1345C,
1337 13
846 13 L846R,
1342 14
1416 5 c.4245+1G>A, c.4245+1G>C, c.4245+2T>A, A1416E, A1416G,
1467 14
1465 11 p.F1465_L1480dup,
1760 11
1761 11 c.5280delG, L1761F, L1761H
1347 15
1710 12 S1710L,
1415 8
932 13
1419 12 K1419E,
1414 12 Q1414H, Q1414H,
931 9
1463 7 N1463Y,
1413 10