Baroudi G, Chahine M
Laval University, Department of Medicine, Canada.
FEBS Lett. 2000 Dec 29;487(2):224-8. doi: 10.1016/s0014-5793(00)02360-7.
Long QT and Brugada syndromes are two hereditary cardiac diseases. Brugada syndrome has so far been associated with only one gene, SCN5A, which encodes the cardiac sodium channel. However, in long QT syndrome (LQTS) at least six genes, including the SCN5A, are implicated. The substitution (D1790G) causes LQTS and the insertion (D1795) induces both LQTS and Brugada syndromes in carrier patients. hH1/insD1795 and hH1/D1790G mutant channels were expressed in the tsA201 human cell line and characterized using the patch clamp technique in whole-cell configuration. Our data revealed a persistent inward sodium current of about 6% at -30 mV for both D1790G and insD1795, and a reduction of 62% of channel expression for the insD1795. Moreover, a shift of steady-state inactivation curve in both mutants was also observed. Our findings uphold the idea that LQT3 is related to a persistent sodium current whereas reduction in the expression level of cardiac sodium channels is one of the biophysical characteristics of Brugada syndrome.
长QT综合征和Brugada综合征是两种遗传性心脏疾病。迄今为止,Brugada综合征仅与一个基因SCN5A相关,该基因编码心脏钠通道。然而,在长QT综合征(LQTS)中,至少有六个基因,包括SCN5A,与之相关。替代突变(D1790G)导致LQTS,插入突变(D1795)在携带患者中诱发LQTS和Brugada综合征。hH1/insD1795和hH1/D1790G突变通道在tsA201人细胞系中表达,并采用全细胞模式的膜片钳技术进行表征。我们的数据显示,对于D1790G和insD1795,在-30 mV时持续内向钠电流约为6%,而insD1795的通道表达减少了62%。此外,在两个突变体中还观察到稳态失活曲线的移位。我们的研究结果支持了这样一种观点,即LQT3与持续钠电流有关,而心脏钠通道表达水平的降低是Brugada综合征的生物物理特征之一。