A Cytoplasmic Domain Mutation in ClC-Kb Affects Long-Distance Communication Across the Membrane
2008

Mutation in ClC-Kb Affects Chloride Channel Function

Sample size: 14 publication Evidence: moderate

Author Information

Author(s): Gilbert Q. Martinez, Merritt Maduke

Primary Institution: Stanford University

Hypothesis

The R538P mutation in ClC-Kb alters its electrophysiological properties due to its proximity to an important membrane-embedded helix.

Conclusion

The R538P mutation in ClC-Kb abolishes calcium activation of the channel, indicating a significant conformational change affecting its function.

Supporting Evidence

  • The R538P mutation leads to Bartter's Syndrome.
  • ClC-Kb-R538P shows altered bromide permeability compared to wild type.
  • Extracellular calcium activation is abolished in ClC-Kb-R538P.

Takeaway

A small change in a protein can stop it from working properly, which can lead to health problems like Bartter's Syndrome.

Methodology

Two-electrode voltage clamp experiments were used to examine the electrophysiological properties of the mutation R538P in both ClC-Kb and ClC-Ka.

Limitations

The study does not definitively establish the mechanism by which the R538P mutation causes Bartter's Syndrome.

Statistical Information

P-Value

p<0.0005

Statistical Significance

p<0.0005

Digital Object Identifier (DOI)

10.1371/journal.pone.0002746

Want to read the original?

Access the complete publication on the publisher's website

View Original Publication