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PubMed Central
Other literature type . 2003
Data sources: PubMed Central
The Journal of Cell Biology
Article . 2003 . Peer-reviewed
Data sources: Crossref
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Dominant cataracts result from incongruous mixing of wild-type lens connexins

Authors: Caterina Sellitto; Francisco J. Martinez-Wittinghan; Thomas W. White; Xiaohua Gong; Leping Li; Richard T. Mathias; Peter R. Brink;

Dominant cataracts result from incongruous mixing of wild-type lens connexins

Abstract

Gap junctions are composed of proteins called connexins (Cx) and facilitate both ionic and biochemical modes of intercellular communication. In the lens, Cx46 and Cx50 provide the gap junctional coupling needed for homeostasis and growth. In mice, deletion of Cx46 produced severe cataracts, whereas knockout of Cx50 resulted in significantly reduced lens growth and milder cataracts. Genetic replacement of Cx50 with Cx46 by knockin rescued clarity but not growth. By mating knockin and knockout mice, we show that heterozygous replacement of Cx50 with Cx46 rescued growth but produced dominant cataracts that resulted from disruption of lens fiber morphology and crystallin precipitation. Impedance measurements revealed normal levels of ionic gap junctional coupling, whereas the passage of fluorescent dyes that mimic biochemical coupling was altered in heterozygous knockin lenses. In addition, double heterozygous knockout lenses retained normal growth and clarity, whereas knockover lenses, where native Cx46 was deleted and homozygously knocked into the Cx50 locus, displayed significantly deficient growth but maintained clarity. Together, these findings suggest that unique biochemical modes of gap junctional communication influence lens clarity and lens growth, and this biochemical coupling is modulated by the connexin composition of the gap junction channels.

Keywords

Male, Mice, Knockout, Heterozygote, Gap Junctions, Cell Communication, Crystallins, Article, Cataract, Connexins, Ion Channels, Disease Models, Animal, Mice, Lens, Crystalline, Mutation, Animals, Female, Eye Proteins, Cell Size, Fluorescent Dyes

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selected citations
These citations are derived from selected sources.
This is an alternative to the "Influence" indicator, which also reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).
BIP!Citations provided by BIP!
popularity
This indicator reflects the "current" impact/attention (the "hype") of an article in the research community at large, based on the underlying citation network.
BIP!Popularity provided by BIP!
influence
This indicator reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).
BIP!Influence provided by BIP!
impulse
This indicator reflects the initial momentum of an article directly after its publication, based on the underlying citation network.
BIP!Impulse provided by BIP!
54
Top 10%
Top 10%
Top 10%
Green
bronze