PUBLICATION

Osterix/sp7 limits cranial bone initiation sites and is required for formation of sutures

Authors
Kague, E., Roy, P., Asselin, G., Hu, G., Stanley, A., Albertson, C., Simonet, J., Fisher, S.
ID
ZDB-PUB-160320-10
Date
2016
Source
Developmental Biology   413(2): 160-72 (Journal)
Registered Authors
Albertson, R. Craig, Fisher, Shannon, Kague, Erika
Keywords
Craniofacial skeleton, Osteogenesis, Osterix/sp7, Sutures, Zebrafish mutant
MeSH Terms
  • Body Patterning
  • Transcriptome
  • Mutation
  • Signal Transduction
  • Humans
  • Zebrafish
  • Transcription Factors/genetics
  • Transcription Factors/physiology*
  • Osteoblasts/cytology
  • Animals
  • Cartilage/embryology
  • Bone Morphogenetic Proteins/metabolism
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/physiology*
  • Cranial Sutures/embryology*
  • Osteogenesis*/genetics
  • Skull/embryology
(all 17)
PubMed
26992365 Full text @ Dev. Biol.
Abstract
During growth, individual skull bones overlap at sutures, where osteoblast differentiation and bone deposition occur. Mutations causing skull malformations have revealed some required genes, but many aspects of suture regulation remain poorly understood. We describe a zebrafish mutation in osterix/sp7, which causes a generalized delay in osteoblast maturation. While most of the skeleton is patterned normally, mutants have specific defects in the anterior skull and upper jaw, and the top of the skull comprises a random mosaic of bones derived from individual initiation sites. Osteoblasts at the edges of the bones are highly proliferative and fail to differentiate, consistent with global changes in gene expression. We propose that signals from the bone itself are required for orderly recruitment of precursor cells and growth along the edges. The delay in bone maturation caused by loss of Sp7 leads to unregulated bone formation, revealing a new mechanism for patterning the skull and sutures.
Genes / Markers
Figures
Figure Gallery (8 images)
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
hu2790
    Point Mutation
    zf195TgTransgenic Insertion
      zf259TgTransgenic Insertion
        zf604TgTransgenic Insertion
          1 - 4 of 4
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          Human Disease / Model
          No data available
          Sequence Targeting Reagents
          No data available
          Fish
          Antibodies
          Name Type Antigen Genes Isotypes Host Organism
          Ab1-smadpolyclonalIgGRabbit
          Ab5-pcnapolyclonal
            IgGRabbit
            Ab8-h3polyclonal
              IgGRabbit
              1 - 3 of 3
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              Orthology
              Gene Orthology
              sp7
              1 - 1 of 1
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              Engineered Foreign Genes
              Marker Marker Type Name
              EGFPEFGEGFP
              mCherryEFGmCherry
              1 - 2 of 2
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              Mapping
              No data available