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Brother of cdo (umleitung) is cell-autonomously required for Hedgehog-mediated ventral CNS patterning in the zebrafish.

The transmembrane protein Brother of Cdo (Boc) has been implicated in Shh-mediated commissural axon guidance, and can both positively and negatively regulate Hedgehog (Hh) target gene transcription, however, little is known about in vivo requirements for Boc during vertebrate embryogenesis. The zebrafish umleitung (uml(ty54)) mutant was identified by defects in retinotectal axon projections. Here, we show that the uml locus encodes Boc and that Boc function is cell-autonomously required for Hh-mediated neural patterning. Our phenotypic analysis suggests that Boc is required as a positive regulator of Hh signaling in the spinal cord, hypothalamus, pituitary, somites and upper jaw, but that Boc might negatively regulate Hh signals in the lower jaw. This study reveals a role for Boc in ventral CNS cells that receive high levels of Hh and uncovers previously unknown roles for Boc in vertebrate embryogenesis.

Pubmed ID: 21115611


  • Bergeron SA
  • Tyurina OV
  • Miller E
  • Bagas A
  • Karlstrom RO


Development (Cambridge, England)

Publication Data

January 8, 2011

Associated Grants

  • Agency: NICHD NIH HHS, Id: HD044929
  • Agency: NINDS NIH HHS, Id: NS039994
  • Agency: NIMH NIH HHS, Id: T32 MH020051

Mesh Terms

  • Animals
  • Axons
  • Central Nervous System
  • Codon, Nonsense
  • Genotype
  • Hedgehog Proteins
  • Immunohistochemistry
  • In Situ Hybridization
  • Pituitary Gland
  • Prosencephalon
  • Signal Transduction
  • Zebrafish
  • Zebrafish Proteins