PUBLICATION

Zebrafish spinal cord oligodendrocyte formation requires boc function

Authors
Kearns, C.A., Walker, M., Ravanelli, A.M., Scott, K., Arzbecker, M.R., Appel, B.
ID
ZDB-PUB-210601-15
Date
2021
Source
Genetics   218(4): (Journal)
Registered Authors
Appel, Bruce, Kearns, Christina, Ravanelli, Andrew M.
Keywords
Sonic hedgehog, glia, myelin, neural progenitors, oligodendrocyte precursor cells
MeSH Terms
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
  • Neural Stem Cells/cytology
  • Neural Stem Cells/metabolism
  • Neurogenesis*
  • Animals
  • Spinal Cord/cytology
  • Spinal Cord/embryology
  • Spinal Cord/metabolism*
  • Oligodendroglia/cytology
  • Oligodendroglia/metabolism*
  • Neural Cell Adhesion Molecules/genetics
  • Neural Cell Adhesion Molecules/metabolism*
  • Zebrafish
(all 14)
PubMed
34057474 Full text @ Genetics
Abstract
The axis of the vertebrate neural tube is patterned, in part, by a ventral to dorsal gradient of Shh signaling. In the ventral spinal cord, Shh induces concentration-dependent expression of transcription factors, subdividing neural progenitors into distinct domains that subsequently produce distinct neuronal and glial subtypes. In particular, progenitors of the pMN domain express the bHLH transcription factor Olig2 and produce motor neurons followed by oligodendrocytes, the myelinating glial cell type of the central nervous system. In addition to its role in patterning ventral progenitors, Shh signaling must be maintained through development to specify pMN progenitors for oligodendrocyte fate. Using a forward genetic screen in zebrafish for mutations that disrupt development of oligodendrocytes, we identified a new mutant allele of boc, which encodes a type I transmembrane protein that functions as a coreceptor for Shh. Embryos homozygous for the bocco25 allele, which creates a missense mutation in a Fibronectin type III domain that binds Shh, have normally patterned spinal cords but fail to maintain pMN progenitors, resulting in a deficit of oligodendrocytes. Using a sensitive fluorescent detection method for in situ RNA hybridization, we found that spinal cord cells express boc in a graded fashion that is inverse to the gradient of Shh signaling activity and that boc function is necessary to maintain pMN progenitors by shaping the Shh signaling gradient.
Genes / Markers
Figures
No images available
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
co24
    		Unknown
    st1001TgTransgenic Insertion
      ty54z
        		Point Mutation
        vu12TgTransgenic Insertion
          1 - 4 of 4
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          Human Disease / Model
          No data available
          Sequence Targeting Reagents
          Fish
          Antibodies
          Orthology
          Engineered Foreign Genes
          Marker Marker Type Name
          EGFPEFGEGFP
          mCherryEFGmCherry
          1 - 2 of 2
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          Mapping
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          Citation
          Kearns, C.A., Walker, M., Ravanelli, A.M., Scott, K., Arzbecker, M.R., Appel, B. (2021) Zebrafish spinal cord oligodendrocyte formation requires boc function. Genetics. 218(4):.