title: A vertebrate-specific and essential role for osterix in osteogenesis revealed by gene knockout in the teleost medaka.
abstract: osterix (osx; sp7) encodes a zinc-finger transcription factor that controls osteoblast differentiation in mammals. Although identified in all vertebrate lineages, its role in non-mammalian bone formation remains elusive. Here, we show that an osx mutation in medaka results in severe bone defects and larval lethality. Pre-osteoblasts fail to differentiate leading to severe intramembranous and perichondral ossification defects. The notochord sheath mineralizes normally, supporting the idea of an osteoblast-independent mechanism for teleost vertebral centra formation. This study establishes a key role for Osx for bone formation in a non-mammalian species, and reveals conserved and non-conserved features in vertebrate bone formation.
authors.affiliation: Department of Biological Sciences, National University of Singapore, Singapore 117543, Singapore.|Centre for Bioimaging Sciences (CBIS), National University of Singapore, Singapore 117543, Singapore.
authors.forename: Tingsheng
authors.initials: T
authors.lastname: Yu
authors.affiliation: Department of Biological Sciences, National University of Singapore, Singapore 117543, Singapore.|Centre for Bioimaging Sciences (CBIS), National University of Singapore, Singapore 117543, Singapore.
authors.forename: Martin
authors.initials: M
authors.lastname: Graf
authors.affiliation: Department of Biological Sciences, National University of Singapore, Singapore 117543, Singapore.|Centre for Bioimaging Sciences (CBIS), National University of Singapore, Singapore 117543, Singapore.
authors.forename: Joerg
authors.initials: J
authors.lastname: Renn
authors.affiliation: Department of Physiological Chemistry, Biocenter, University of Würzburg, and Comprehensive Cancer Center Mainfranken, University Clinic Würzburg, Würzburg 97080, Germany.|Texas Institute for Advanced Study and Department of Biology, Texas A&M University, College Station, TX 77843, USA.
authors.forename: Manfred
authors.initials: M
authors.lastname: Schartl
authors.affiliation: Department of Biology, Research Group Evolutionary Developmental Biology, Ghent University, Ghent B-9000, Belgium.
authors.forename: Daria
authors.initials: D
authors.lastname: Larionova
authors.affiliation: Department of Biology, Research Group Evolutionary Developmental Biology, Ghent University, Ghent B-9000, Belgium.
authors.forename: Ann
authors.initials: A
authors.lastname: Huysseune
authors.affiliation: Department of Biology, Research Group Evolutionary Developmental Biology, Ghent University, Ghent B-9000, Belgium.
authors.forename: Paul Eckhard
authors.initials: PE
authors.lastname: Witten
authors.affiliation: Department of Biological Sciences, National University of Singapore, Singapore 117543, Singapore dbswcw@nus.edu.sg.|Centre for Bioimaging Sciences (CBIS), National University of Singapore, Singapore 117543, Singapore.
authors.forename: Christoph
authors.identifier: 0000-0002-0330-6692
authors.initials: C
authors.lastname: Winkler
mesh_terms: D000818:Animals; D030801:Animals, Genetically Modified; D002113:Calcification, Physiologic / Q000235:genetics; D018507:Gene Expression Regulation, Developmental; D055786:Gene Knockout Techniques; D009672:Notochord / Q000196:embryology; D009990:Oryzias / Q000196:embryology* / Q000235:genetics*; D010012:Osteogenesis / Q000235:genetics*; D010802:Phylogeny; D000076182:Sp7 Transcription Factor; D013045:Species Specificity; D014157:Transcription Factors / Q000235:genetics / Q000502:physiology*; D014714:Vertebrates / Q000196:embryology / Q000235:genetics; D029961:Zebrafish Proteins / Q000502:physiology
chemical_list: D000076182:Sp7 Transcription Factor; D014157:Transcription Factors; D029961:Zebrafish Proteins; C541742:sp7 protein, zebrafish
keywords: Bone modelling; Medaka; Osteoblasts; Osteogenesis; Skeleton