title: Identification of adult mineralized tissue zebrafish mutants.
abstract: Zebrafish craniofacial, skeletal, and tooth development closely resembles that of higher vertebrates. Our goal is to identify viable adult zebrafish mutants that can be used as models for human mineralized craniofacial, dental, and skeletal system disorders. We used a large-scale forward-genetic chemical N-ethyl-nitroso-urea mutagenesis screen to identify 17 early lethal homozygous recessive mutants with defects in craniofacial cartilage elements, and 7 adult homozygous recessive mutants with mineralized tissue phenotypes including craniofacial shape defects, fused sutures, dysmorphic or missing skeletal elements, scoliosis, and neural arch defects. One mutant displayed both an early lethal homozygous phenotype and an adult heterozygous phenotype. These results extend the utility of the zebrafish model beyond the embryo to study human bone and cartilage disorders.
authors.affiliation: Divison of Craniofacial and Molecular Genetics, Department of Oral and Maxillofacial Pathology, Tufts University, Boston, MA 02111, USA.
authors.forename: Viktoria
authors.initials: V
authors.lastname: Andreeva
authors.forename: Michelle H
authors.initials: MH
authors.lastname: Connolly
authors.forename: Caitlin
authors.initials: C
authors.lastname: Stewart-Swift
authors.forename: Daniel
authors.initials: D
authors.lastname: Fraher
authors.forename: Jeffrey
authors.initials: J
authors.lastname: Burt
authors.forename: Justin
authors.initials: J
authors.lastname: Cardarelli
authors.forename: Pamela C
authors.initials: PC
authors.lastname: Yelick
mesh_terms: D000423:Alcian Blue; D000818:Animals; D000880:Anthraquinones; D002356:Cartilage / Q000002:abnormalities*; D019465:Craniofacial Abnormalities / Q000235:genetics*; D004195:Disease Models, Animal; D005038:Ethylnitrosourea; D005808:Genes, Recessive / Q000235:genetics; D016296:Mutagenesis; D010641:Phenotype; D015027:Zebrafish
chemical_list: D000880:Anthraquinones; C010078:alizarin; D000423:Alcian Blue; D005038:Ethylnitrosourea