SCN5A Variant L1761H

Summary of observed carriers, functional annotations, and structural context for SCN5A L1761H. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

61%

4/12 effective observations

Estimated BrS1 penetrance

20%

2/12 effective observations

Total carriers

2

0 BrS1 · 2 LQT3 · 0 unaffected

L1761H 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 (%)
-6.47 1 -4.22 0.989 26 61

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
20197117 2010 2 1 0 1 AV block
19716085 2009 1 1 0 0
Literature, cohort, and gnomAD 2 0 2 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)
19716085 2009
20197117 2010

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 L1761H.
Neighbour residue Distance (Å) Observed variants
1417 9
1765 6
1340 14 V1340I,
1457 12
1757 5
1413 11
1472 14 N1472S, p.N1472del,
1339 15 p.L1339del, L1339F,
1756 6 I1756V,
1461 10 T1461S, T1461S,
1764 8 V1764F, c.5290delG,
371 15 Q371E,
1333 12
1711 14 c.5131delG,
1754 10
1707 12
1458 11 S1458Y,
1671 14
935 14 L935P,
1762 5 p.I1762del, I1762M,
1470 13
1464 10 c.4389_4396delCCTCTTTA, L1464P,
1466 9 c.4396_4397insG,
1753 10 T1753A,
1767 13 Y1767C,
1660 14 I1660V, I1660S,
1329 15 G1329S,
1769 13
1418 13
402 14 F402L, F402L, F402L,
1766 10 M1766L, M1766T, M1766V, M1766L,
1768 11 I1768V,
1473 15 F1473S, F1473C,
1334 9 I1334V,
1341 11
1468 11 V1468F, V1468A,
1663 14
1462 7
938 15
1759 7 S1759C,
1327 12
1709 11 T1709M, T1709R, p.T1709del,
1758 7 I1758V, p.I1758del,
1459 14 c.4376_4379delTCTT,
1755 9
1330 12 A1330P, A1330D, A1330T,
1460 14 F1460L, F1460L, F1460L,
1713 11
1338 9 L1338V,
1708 11 T1708I,
1409 14 Y1409C, Y1409X,
1345 12 W1345C, W1345C,
1337 10
1342 14
1326 14 A1326S,
1763 8 V1763L, V1763L, V1763M,
1751 15
1416 11 c.4245+1G>C, c.4245+2T>A, A1416E, c.4245+1G>A, A1416G,
1332 14 P1332L, P1332Q,
1465 5 p.F1465_L1480dup,
1760 4
1467 11
1752 13
1331 11 I1331V,
1761 0 L1761H, L1761F, c.5280delG,
1469 9 I1469V,
1336 14
1710 10 S1710L,
1415 15
1335 13 M1335R,
1419 14 K1419E,
1667 13 V1667I,
1414 13 Q1414H, Q1414H,
1664 13
1463 9 N1463Y,