ZFIN ID: ZDB-PUB-050119-9
Mutations in connexin43 (GJA1) perturb bone growth in zebrafish fins
Iovine, M.K., Higgins, E.P., Hindes, A., Coblitz, B., and Johnson, S.L.
Date: 2005
Source: Developmental Biology   278(1): 208-219 (Journal)
Registered Authors: Higgins, Emmett, Hindes, Anna, Iovine, M. Kathryn, Johnson, Stephen L.
Keywords: Fin growth; connexin43; Gap junctions; short fin; Zebrafish; Bone growth
MeSH Terms:
  • Alleles
  • Animals
  • Base Sequence
  • Bone Development/genetics*
  • Bone Regeneration/genetics
  • Chromosome Mapping
  • Connexin 43/genetics*
  • Connexin 43/physiology
  • DNA/genetics
  • Female
  • Gene Expression Regulation, Developmental
  • Male
  • Models, Biological
  • Mutation*
  • Phenotype
  • Zebrafish/genetics*
  • Zebrafish/growth & development*
  • Zebrafish/physiology
  • Zebrafish Proteins/genetics*
PubMed: 15649473 Full text @ Dev. Biol.
Mechanisms that regulate the size and shape of bony structures are largely unknown. The molecular identification of the fin length mutant short fin (sof), which causes defects in the length of bony fin ray segments, may provide insights regarding the regulation of bone growth. In this report, we demonstrate that the sof phenotype is caused by mutations in the connexin43 (cx43) gene. This conclusion is supported by genetic mapping, reduced expression of cx43 in the original sof allele (sof(b123)), identification of missense mutations in three ENU-induced alleles, and by demonstration of partially abrogated cx43 function in sof(b123) embryos. Expression of cx43 was identified in cells flanking the germinal region of newly growing segments as well as in the osteoblasts at segment boundaries. This pattern of cx43 expression in cells lateral to new segment growth is consistent with a model where cx43-expressing cells represent a biological ruler that measures segment size. This report identifies the first gene identification for a fin length mutation (sof) as well as the first connexin mutations in zebrafish, and therefore reveals a critical role for local cell-cell communication in the regulation of bone size and growth.