Barttin is a Cl- channel β-subunit crucial for renal Cl- reabsorption and inner ear K+ secretion
doi: 10.1038/35107099
pmid: 11734858
Barttin is a Cl- channel β-subunit crucial for renal Cl- reabsorption and inner ear K+ secretion
Renal salt loss in Bartter's syndrome is caused by impaired transepithelial transport in the loop of Henle. Sodium chloride is taken up apically by the combined activity of NKCC2 (Na+-K--2Cl- cotransporters) and ROMK potassium channels. Chloride ions exit from the cell through basolateral ClC-Kb chloride channels. Mutations in the three corresponding genes have been identified that correspond to Bartter's syndrome types 1-3. The gene encoding the integral membrane protein barttin is mutated in a form of Bartter's syndrome that is associated with congenital deafness and renal failure. Here we show that barttin acts as an essential beta-subunit for ClC-Ka and ClC-Kb chloride channels, with which it colocalizes in basolateral membranes of renal tubules and of potassium-secreting epithelia of the inner ear. Disease-causing mutations in either ClC-Kb or barttin compromise currents through heteromeric channels. Currents can be stimulated further by mutating a proline-tyrosine (PY) motif on barttin. This work describes the first known beta-subunit for CLC chloride channels and reveals that heteromers formed by ClC-K and barttin are crucial for renal salt reabsorption and potassium recycling in the inner ear.
- Universitäts-Kinderklinik Würzburg Germany
- University of Freiburg Germany
- Universität Hamburg Germany
- University Hospital Würzburg Germany
Xenopus, Anion Transport Proteins, Bartter Syndrome, Membrane Proteins, Kidney, Recombinant Proteins, Absorption, Cell Line, Protein Subunits, Kidney Tubules, Chlorides, Chloride Channels, Ear, Inner, Potassium, Animals
Xenopus, Anion Transport Proteins, Bartter Syndrome, Membrane Proteins, Kidney, Recombinant Proteins, Absorption, Cell Line, Protein Subunits, Kidney Tubules, Chlorides, Chloride Channels, Ear, Inner, Potassium, Animals
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