Zebrafish short fin mutations in connexin43 lead to aberrant gap junctional intercellular communication.
Mutations in the zebrafish connexin43 (cx43) gene cause the short fin phenotype, indicating that direct cell-cell communication contributes to bone length. Three independently generated cx43 alleles exhibit short segments of variable sizes, suggesting that gap junctional intercellular communication may regulate bone growth. Dye coupling assays showed that all alleles are capable of forming gap junction channels. However, ionic coupling assays revealed allele-specific differences in coupling efficiency and gating. For instance, oocyte pairs expressing the weakest allele exhibited much higher levels of coupling than either of the strong alleles. Therefore, measurable differences in Cx43 function may be correlated with the severity of defects in bone length.
Hoptak-Solga
Angela D
AD
Lehigh University, Department of Biological Sciences, Iacocca B-217, Bethlehem, PA 18015, USA.
Klein
Kathryn A
KA
DeRosa
Adam M
AM
White
Thomas W
TW
Iovine
M Kathryn
MK
D000818:Animals; D050281:Bones of Lower Extremity / Q000002:abnormalities; D002450:Cell Communication / Q000235:genetics*; D018031:Connexin 43 / Q000235:genetics*; D017629:Gap Junctions / Q000235:genetics*; D006367:HeLa Cells; D006801:Humans; D017880:Limb Deformities, Congenital / Q000235:genetics*; D020125:Mutation, Missense / Q000502:physiology; D018408:Patch-Clamp Techniques; D010641:Phenotype; D014162:Transfection; D015027:Zebrafish / Q000235:genetics*; D029961:Zebrafish Proteins / Q000235:genetics*
D018031:Connexin 43; D029961:Zebrafish Proteins