Key result
SCN5A mutations were found significantly more often in familial Brugada syndrome (38%) compared to sporadic cases (0%) (p=0.001).
Why the study?
Does the incidence of SCN5A mutations differ between familial and sporadic Brugada syndrome?
Observational (n=44)
Yes
Does the incidence of SCN5A mutations differ between familial and sporadic Brugada syndrome?
Absolute Event Rate: 38% vs 0%
p-value: p=0.001
SCN5A mutations are significantly more common in familial Brugada syndrome compared to sporadic cases, suggesting genetic testing is most useful in familial disease.
SCN5A mutations associate more with familial than sporadic Brugada syndrome; supports targeted testing hypotheses but leaves open prospective validation before practice change.
The Brugada syndrome (BS) is a distinct form of idiopathic ventricular fibrillation and may cause sudden cardiac death in healthy young individuals. In the surface ECG, BS can be recognized by an atypical right bundle branch block and ST-segment elevation in the right precordial leads. Mutations in the cardiac sodium channel gene SCN5A are only known to cause BS. In a multi-center effort, we have collected clinical data on 44 unrelated index patients and family members and performed a complete genetic analysis of SCN5A. In 37% the disease was familial, whereas in the majority it was sporadic (63%). Five novel SCN5A mutations (2602delC, resulting in: E867X; 2581_2582del TT: F861fs951X; 2673G>A: E1225K; 4435_4437delAAG: K1479del; and 5425C>A: S1812X) were found and were randomly located in SCN5A. Mutation frequencies (SCN5A+) differed significantly between familial (38%) and sporadic disease (0%) (p=0.001). Disease penetrance was complete in the SCN5A+ adult patients, but incomplete in SCN5A+ children (17%). Genetic testing of SCN5A is especially useful in familial disease to identify individuals at cardiac risk. In sporadic cases, however, a genetic basis and the value of mutation screening has to be further determined. These results are in line with a possibly genetic and clinical heterogeneity of BS.
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Schulze‐Bahr et al. (2003) conducted an observational in Brugada syndrome (n=44). SCN5A genetic analysis vs. Familial vs sporadic disease was evaluated on SCN5A mutation frequency (p=0.001). SCN5A mutations were found significantly more often in familial Brugada syndrome (38%) compared to sporadic cases (0%) (p=0.001).
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