SCN5A Variant D1243N

Summary of observed carriers, functional annotations, and structural context for SCN5A D1243N. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

1%

0/57 effective observations

Estimated BrS1 penetrance

10%

5/57 effective observations

Total carriers

47

5 BrS1 · 0 LQT3 · 42 unaffected

D1243N is present in 41 alleles 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 0 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 (%)
-4.89 1 4.97 0.924 11 0

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
22885917 2012 2 0 2 0
26173111 2015 1 0 1 0
26921764 2016 2 0 2 0
26746457 2016 1 0 0 0
20129283 2010 1 0 1 0
20129283 2010 1 0 1 0
20129283 2010 5 0 5 0
29325976 2018 3 0 3 0
Literature, cohort, and gnomAD 47 42 0 5
Variant features alone 15 15 0 0

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)
22885917 2012
26173111 2015
26921764 2016
26746457 2016
20129283 2010
20129283 2010
20129283 2010
29325976 2018
32533946 2020 HEK 115 5.7 7.8

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 D1243N.
Neighbour residue Distance (Å) Observed variants
1245 7 M1245I, M1245I, M1245I,
1218 10 S1218T, S1218I,
1281 10 c.3840+1G>A, V1281F,
1304 11 T1304M,
1217 13
1243 0 D1243N,
1285 7
1299 11 c.3894delC,
1220 13 G1220E,
1673 15
1298 15 P1298L,
1283 10 L1283M,
1241 8
1309 14 R1309C, R1309H
1226 15
1288 11 A1288G,
1221 11 A1221V,
1242 5
1219 12 S1219N,
1251 12 V1251M,
1279 11 V1279I,
1239 7 L1239P,
1306 7 R1306S, R1306H,
1244 6 K1244E,
1286 8
1305 12
1282 7 S1282A,
1246 7
1235 12
1302 10 p.L1302Vfs18,
1247 6 T1247I,
1237 11 V1237F,
1307 13
1289 9
1223 15 c.3667delG,
1222 10 p.L1222LfsX7, L1222R,
1300 14
1229 14
1215 13 I1215V,
1301 11
1214 13 M1214T,
1253 15 E1253G,
1284 10
1280 13
1250 9
1240 5 E1240Q,
1248 9
1225 11 E1225K, G1225K,
1287 12
1290 14
1278 11 I1278N,
1238 10
1236 12 K1236R, K1236N, K1236N,
1249 10 V1249D,
1303 6 R1303W, R1303Q,