Abstract

Polycystin-2, the product of the human PKD2 gene, whose mutations cause autosomal dominant polycystic kidney disease, is a large conductance, Ca(2+)-permeable non-selective cation channel. Polycystin-2 is functionally expressed in the apical membrane of the human syncytiotrophoblast, where it may play a role in the control of fetal electrolyte homeostasis. Little is known, however, about the mechanisms that regulate polycystin-2 channel function. In this study, the role of pH in the regulation of polycystin-2 was assessed by ion channel reconstitution of both apical membranes of human syncytiotrophoblast and the purified FLAG-tagged protein from in vitro transcribed/translated material. A kinetic analysis of single channel currents, including dwell time histograms, confirmed two open and two close states for spontaneous channel behavior and a strong voltage dependence of the open probability of the channel (P(o)). A reduction of cis pH (pH(cis)) decreased P(o) and shifted the voltage dependence of channel function but had no effect on the single channel conductance. An increase in pH(cis), in contrast, increased NP(o) (channel number times P(o)). Elimination of the H(+) chemical gradient did not reverse the low pH(cis) inhibition of polycystin-2. Similar findings confirmed the pH effect on the in vitro translated, FLAG-tagged purified polycystin-2. The data indicate the presence of an H(+) ion regulatory site in the channel protein, which is accessible from the cytoplasmic side of the protein. This protonation site controls polycystin-2 cation-selective channel activity.

Highlights

  • Autosomal dominant polycystic kidney disease (ADPKD)1 is a prevalent human genetic disorder affecting 1:400 to 1:1000 individuals worldwide

  • Kinetic Analysis of Single Channel Currents of hST Polycystin-2—Human syncytiotrophoblast apical membranes were reconstituted in the presence of asymmetrical KCl (150 mM) in the cis side and 15 mM Kϩ in the trans side

  • The present study demonstrates that polycystin-2 cation channel function is voltage-dependent and highly sensitive to changes in pH

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Summary

Introduction

Autosomal dominant polycystic kidney disease (ADPKD)1 is a prevalent human genetic disorder affecting 1:400 to 1:1000 individuals worldwide. Human polycystin-2 from two different sources, including hST apical membranes and the in vitro transcribed/transduced human gene product, was functionally reconstituted in a lipid bilayer system where the effect(s) of pH on its cation channel activity was determined.

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