SCN5A Variant c.2582_2583delTT

Summary of observed carriers, functional annotations, and structural context for SCN5A c.2582_2583delTT. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

10%

1/21 effective observations

Estimated BrS1 penetrance

70%

14/21 effective observations

Total carriers

11

11 BrS1 · 0 LQT3 · 0 unaffected

c.2582_2583delTT has not been reported in gnomAD. This residue resides in a Hotspot region for Brugada syndrome and a Mild_Hotspot region for LQT3.

Variant features alone are equivalent to phenotyping 3 individuals for Brugada syndrome and 1 individuals for LQT3.

In silico predictors

Variant-level computational predictors.
PROVEAN PolyPhen-2 BLAST-PSSM REVEL Penetrance Density BrS (%) Penetrance Density LQT3 (%)
NA NA NA None 51 39

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
19251209 2009 1 0 1 0
19029124 2009 1 0 1 0
21273195 2011 7 0 7 0
22885917 2012 3 0 3 0
26921764 2016 1 0 1 0
26941339 2016 2 0 2 0
20129283 2010 11 0 11 0
29759671 2018 1 0 1 0
Literature, cohort, and gnomAD 11 0 0 11
Variant features alone 15 11 1 3

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)
19251209 2009
19029124 2009
21273195 2011
22885917 2012
26921764 2016
26941339 2016
20129283 2010
29759671 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 c.2582_2583delTT.
Neighbour residue Distance (Å) Observed variants
891 11 I891N, I891T,
880 11
888 15
223 14 V223L,
856 8 V856L, V856L,
890 10 I890T,
901 15 E901K, S901L,
919 9
862 5
867 12 E867K, E867Q, E867X,
859 8
894 14 I894M,
863 8
904 14 W904X,
887 10
864 7
886 13 H886P, H886Q, H886Q,
216 11 S216X, S216L,
221 12
909 9
852 14
854 10 c.2559delT,
876 14
857 5 G857D,
868 13 c.2602delC, L868X,
902 10
882 11
881 6
921 13
922 13 V922I
860 4 p.L860fsx89,
911 10 G911E,
920 14
889 15
858 8 M858L, M858L,
217 12
918 8
855 10
917 11 L917V, L917R,
865 7
913 9
916 11
912 12 Q912R,
884 14
906 6
866 12 S866P, S866L,
910 6 S910L,
903 11 p.M903CfsX29,
853 11
219 13 p.R219HfsX11, R219C, c.656_657insATTCA, R219H,
877 13
879 15 W879R, W879R,
883 13
905 11
915 6 C915R,
215 13 p.L215CfsX10,
850 15 V850M, c.2549_2550insTG,
908 12
914 7
861 0 p.F861WfsX90, c.2582_2583delTT,
220 9 T220I,
907 10