Mutations affecting development of the notochord in zebrafish
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Updated: Nov 3, 2026

Shifting Zebrafish Lethal Skeletal Mutant Penetrance by Progeny Testing
Published on: September 1, 2017
D L Stemple1, L Solnica-Krezel, F Zwartkruis
1Cardiovascular Research Center, Massachusetts General Hospital, Charlestown 02129, USA.
Abstract:
The notochord is critical for the normal development of vertebrate embryos. It serves both as the major skeletal element of the embryo and as a signaling source for the establishment of pattern within the neurectoderm, the paraxial mesoderm and other tissues. In a large-scale systematic screen of mutations affecting embryogenesis in zebrafish we identified 65 mutations that fall into 29 complementation groups, each leading to a defect in the formation and/or maintenance of the notochord. These mutations produce phenotypic abnormalities at numerous stages of notochord development, thereby establishing a phenotypic pathway, which in turn suggests a genetic pathway for the development of the notochord. Perturbations within adjacent tissues in mutant embryos further indicate the importance of notochord-derived signals for patterning within the embryo and suggest that these mutations will yield additional insight into the cues that regulate these patterning processes.

