SCN5A Variant C280Y

Summary of observed carriers, functional annotations, and structural context for SCN5A C280Y. Data combine curated literature, international cohorts, and gnomAD observations.

Estimated LQT3 penetrance

8%

0/11 effective observations

Estimated BrS1 penetrance

58%

6/11 effective observations

Total carriers

1

1 BrS1 · 0 LQT3 · 0 unaffected

C280Y has not been reported in gnomAD. This residue resides in a Hotspot region for Brugada syndrome and a Non_Hotspot region for LQT3.

Variant features alone are equivalent to phenotyping 5 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 (%)
-10.65 1 -4.54 0.973 78 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
23321620 2013 1 0 1 0
Literature, cohort, and gnomAD 1 0 0 1
Variant features alone 15 10 0 5

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)
23321620 2013

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 C280Y.
Neighbour residue Distance (Å) Observed variants
328 11
333 11 c.998+1G>A, c.998+5G>A,
277 12
326 5
276 13 L276Q, L276P,
348 12 P348A,
317 15 K317E, K317M, K317N, K317N,
279 5
385 14 A385T,
338 9
278 8 H278D, H278R,
334 9 c.999-424_1338+81del,
332 12 A332T,
343 12
327 8
339 6
384 10 S384T,
284 13
329 14
282 8 R282C, R282H,
340 8 R340W, R340Q,
285 14 T285K,
283 10
341 6 C341Y,
274 14 G274C,
335 7 C335S, C335R, C335S,
319 11 G319S, G319R, G319C,
325 7 L325R,
324 7
321 9 S321Y,
345 12
275 13 N275K, N275K,
383 9
280 0 C280Y,
323 6
347 14
337 12
320 10 T320N,
1689 14 D1689N
342 7
346 14 E346K, E346G, E346X, E346D, E346D,
336 7 P336L,
344 10 A344S,
381 15 c.1140+1G>A, c.1141-3C>A,
322 10
1691 15
380 12
281 5 V281M,