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Updated: Jul 17, 2026
Analysis of Embryonic and Larval Zebrafish Skeletal Myofibers from Dissociated Preparations
Published on: November 13, 2013
Mutations in cdon and boc affect trunk neural crest cell migration and slow-twitch muscle development in zebrafish
Ezra Lencer1,2, Addison Rains2,3, Erin Binne2
1Department of Cell and Developmental Biology, University of Colorado, Anschutz Medical Campus Aurora, CO 80045, USA.
Abstract:
The transmembrane proteins cdon and boc are implicated in regulating hedgehog signaling during vertebrate development. Recent work showing roles for these genes in axon guidance and neural crest cell migration suggest that cdon and boc may play additional functions in regulating directed cell movements. We use newly generated and existing mutants to investigate a role for cdon and boc in zebrafish neural crest cell migration. We find that single mutant embryos exhibit normal neural crest phenotypes, but that neural crest migration is strikingly disrupted in double cdon;boc mutant embryos. We further show that this migration phenotype is associated with defects in the differentiation of slow-twitch muscle cells, and the loss of a Col1a1a-containing extracellular matrix, suggesting that neural crest defects may be a secondary consequence to defects in mesoderm development. Combined, our data add to a growing literature showing that cdon and boc act synergistically to promote hedgehog signaling during vertebrate development, and suggest that the zebrafish can be used to study the function of hedgehog receptor paralogs.
Insights
The transmembrane proteins Cdon and Boc work together to regulate hedgehog signaling. Zebrafish lacking both Cdon and Boc show severe defects in neural crest cell migration and muscle development.
Area of Science:
- Developmental Biology
- Cell Biology
- Genetics
Background:
- Transmembrane proteins Cdon (Cellular distribution and differentiation) and Boc (Brother of Cdon) are known regulators of hedgehog signaling.
- Previous studies suggest roles for Cdon and Boc in axon guidance and neural crest cell migration, indicating potential functions in directed cell movement.
Purpose of the Study:
- To investigate the role of Cdon and Boc in zebrafish neural crest cell migration using genetic mutants.
- To understand the synergistic functions of Cdon and Boc in vertebrate development.
Main Methods:
- Generation and analysis of zebrafish mutants for cdon and boc.
- Phenotypic analysis of neural crest cell migration and muscle differentiation.
- Assessment of extracellular matrix components.
Main Results:
- Single cdon or boc mutant zebrafish embryos display normal neural crest phenotypes.
- Double cdon;boc mutant embryos exhibit significantly disrupted neural crest cell migration.
- The observed neural crest defects correlate with impaired slow-twitch muscle cell differentiation and loss of a Col1a1a-containing extracellular matrix.
Conclusions:
- Cdon and Boc act synergistically to regulate hedgehog signaling during vertebrate development.
- Defects in zebrafish neural crest migration in double mutants may be secondary to mesodermal developmental issues.
- Zebrafish serve as a valuable model for studying the function of hedgehog receptor paralogs.
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