American Journal of Physiology-Heart and Circulatory Physiology · 2010 · 30 citations · 33 references
Life SciencesGeneticsMolecular BiologyElectrophysiologyGene ExpressionMolecular PhysiologyCardiac Ion ChannelsChannelopathiesIon Channel ExpressionIon Channel PhysiologyIon Channel RegulationIon ChannelsHeterologous Expression SystemsIon Channel Expression AlterationIon Channel Expression RegulationIon Channel Surface ExpressionLong Qt Syndrome ElectrophysiologyPotassium Channel RegulationRabbit ModelsTransgenic Cell Lines
We previously reported a transgenic rabbit model of long QT syndrome based on overexpression of pore mutants of repolarizing K(+) channels KvLQT1 (LQT1) and HERG (LQT2).The transgenes in these rabbits eliminated the slow and fast components of the delayed rectifier K(+) current (I(Ks) and I(Kr), respectively), as expected. Interestingly, the expressed pore mutants of HERG and KvLQT1 downregulated the remaining reciprocal repolarizing currents, I(Ks) and I(Kr), without affecting the steady-state levels of the native polypeptides. Here, we sought to further explore the functional interactions between HERG and KvLQT1 in heterologous expression systems. Stable Chinese hamster ovary (CHO) cell lines expressing KvLQT1-minK or HERG were transiently transfected with expression vectors coding for mutant or wild-type HERG or KvLQT1. Transiently expressed pore mutant or wild-type KvLQT1 downregulated I(Kr) in HERG stable CHO cell lines by 70% and 44%, respectively. Immunostaining revealed a severalfold lower surface expression of HERG, which could account for the reduction in I(Kr) upon KvLQT1 expression. Deletion of the KvLQT1 NH(2)-terminus did not abolish the downregulation, suggesting that the interactions between the two channels are mediated through their COOH-termini. Similarly, transiently expressed HERG reduced I(Ks) in KvLQT1-minK stable cells. Coimmunoprecipitations indicated a direct interaction between HERG and KvLQT1, and surface plasmon resonance analysis demonstrated a specific, physical association between the COOH-termini of KvLQT1 and HERG. Here, we present an in vitro model system consistent with the in vivo reciprocal downregulation of repolarizing currents seen in transgenic rabbit models, illustrating the importance of the transfection method when studying heterologous ion channel expression and trafficking. Moreover, our data suggest that interactions between KvLQT1 and HERG are mediated through COOH-termini.
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A molecular basis for cardiac arrhythmia: HERG mutations cause long QT syndrome
Mark Curran, Igor Splawski, Katherine W. Timothy et al. · Cell · 1995 · 2.3K citations · Full text
Coassembly of KVLQT1 and minK (IsK) proteins to form cardiac IKS potassium channel
Michael C. Sanguinetti, Mark Curran, Anruo Zou et al. · Nature · 1996 · 1.8K citations
KvLQT1 and IsK (minK) proteins associate to form the IKS cardiac potassium current
Jacques Barhanin, Florian Lesage, Eric Guillemare et al. · Nature · 1996 · 1.6K citations
Spectrum of Mutations in Long-QT Syndrome Genes
Igor Splawski, Jiaxiang Shen, Katherine W. Timothy et al. · Circulation · 2000 · 1.2K citations · Full text
Life Sciences, Cardiology, Genetics +15