Mutations in cdon and boc affect trunk neural crest cell migration and slow-twitch muscle development in zebrafish

Ezra Lencer, Addison Rains, Erin Binne, Rytis Prekeris, Kristin B. Artinger

Research output: Contribution to journalArticlepeer-review


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.

Original languageEnglish (US)
Article numberdev201304
JournalDevelopment (Cambridge)
Issue number14
StatePublished - Jul 2023

Bibliographical note

Funding Information:
This work was in part supported by the National Institutes of Health (GM122768 to R.P.) and a University of Colorado RNA Biosciences Initiative award to E.L. and K.A. E.L. was supported by National Institutes of Health Ruth L. Kirschstein National Research Service Awards (T32CA17468 and F32HD103406). Deposited in PMC for release after 12 months.

Publisher Copyright:
© 2023. Published by The Company of Biologists Ltd.


  • Adaxial cell
  • Cell guidance
  • Cell migration
  • Extracellular matrix
  • Hedgehog signaling
  • Neural crest cell

PubMed: MeSH publication types

  • Journal Article
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't


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