Restier Lioara, Cheng Lan, Sanguinetti Michael C
Nora Eccles Harrison Cardiovascular Research & Training Institute and Department of Physiology, University of Utah, Salt Lake City, UT 84112, USA.
J Physiol. 2008 Sep 1;586(17):4179-91. doi: 10.1113/jphysiol.2008.157511. Epub 2008 Jul 3.
The slow delayed rectifier K(+) current (I(Ks)) is a major determinant of action potential repolarization in the heart. I(Ks) channels are formed by coassembly of pore-forming KCNQ1 alpha-subunits and ancillary KCNE1 beta-subunits. Two gain of function mutations in KCNQ1 subunits (S140G and V141M) have been associated with atrial fibrillation (AF). Previous heterologous expression studies found that both mutations caused I(Ks) to be instantaneously activated, presumably by preventing channel closure. The purpose of this study was to refine our understanding of the channel gating defects caused by these two mutations located in the S1 domain of KCNQ1. Site-directed mutagenesis was used to replace S140 or V141 with several other natural amino acids. Wild-type and mutant channels were heterologously expressed in Xenopus oocytes and channel function was assessed with the two-microelectrode voltage clamp technique. Long intervals between voltage clamp pulses revealed that S140G and V141M KCNQ1-KCNE1 channels are not constitutively active as previously reported, but instead exhibit extremely slow deactivation. The slow component of I(Ks) deactivation was decreased 62-fold by S140G and 140-fold by the V141M mutation. In addition, the half-point for activation of these mutant I(Ks) channels was approximately 50 mV more negative than wild-type channels. Other substitutions of S140 or V141 in KCNQ1 caused variable shifts in the voltage dependence of activation, but slowed I(Ks) deactivation to a much lesser extent than the AF-associated mutations. Based on a published structural model of KCNQ1, S140 and V141 are located near E160 in S2 and R237 in S4, two charged residues that could form a salt bridge when the channel is in the open state. In support of this model, mutational exchange of E160 and R237 residues produced a constitutively open channel. Together our findings suggest that altered charge-pair interactions within the voltage sensor module of KCNQ1 subunits may account for slowed I(Ks) deactivation induced by S140 or V141.
缓慢延迟整流钾电流(I(Ks))是心脏动作电位复极化的主要决定因素。I(Ks)通道由孔形成亚基KCNQ1α亚基和辅助亚基KCNE1β亚基共同组装而成。KCNQ1亚基中的两个功能获得性突变(S140G和V141M)与心房颤动(AF)相关。先前的异源表达研究发现,这两个突变均导致I(Ks)瞬间激活,推测是通过阻止通道关闭实现的。本研究的目的是进一步了解由位于KCNQ1 S1结构域的这两个突变引起的通道门控缺陷。采用定点诱变将S140或V141替换为其他几种天然氨基酸。野生型和突变型通道在非洲爪蟾卵母细胞中进行异源表达,并使用双微电极电压钳技术评估通道功能。电压钳脉冲之间的长时间间隔显示,S140G和V141M KCNQ1-KCNE1通道并非如先前报道的那样持续激活,而是表现出极其缓慢的失活。S140G使I(Ks)失活的慢成分降低了62倍,V141M突变使其降低了140倍。此外,这些突变型I(Ks)通道的激活中点比野生型通道负约50 mV。KCNQ1中S140或V141的其他替换导致激活电压依赖性发生不同程度的偏移,但使I(Ks)失活减慢的程度远小于与AF相关的突变。基于已发表的KCNQ1结构模型,S140和V141位于S2中的E160和S4中的R237附近,这两个带电残基在通道处于开放状态时可形成盐桥。支持该模型的是,E160和R237残基的突变交换产生了一个持续开放的通道。我们的研究结果共同表明,KCNQ1亚基电压传感器模块内电荷对相互作用的改变可能是S140或V141诱导I(Ks)失活减慢的原因。