title: Small teleost fish provide new insights into human skeletal diseases.
abstract: Small teleost fish such as zebrafish and medaka are increasingly studied as models for human skeletal diseases. Efficient new genome editing tools combined with advances in the analysis of skeletal phenotypes provide new insights into fundamental processes of skeletal development. The skeleton among vertebrates is a highly conserved organ system, but teleost fish and mammals have evolved unique traits or have lost particular skeletal elements in each lineage. Several unique features of the skeleton relate to the extremely small size of early fish embryos and the small size of adult fish used as models. A detailed analysis of the plethora of interesting skeletal phenotypes in zebrafish and medaka pushes available skeletal imaging techniques to their respective limits and promotes the development of new imaging techniques. Impressive numbers of zebrafish and medaka mutants with interesting skeletal phenotypes have been characterized, complemented by transgenic zebrafish and medaka lines. The advent of efficient genome editing tools, such as TALEN and CRISPR/Cas9, allows to introduce targeted deficiencies in genes of model teleosts to generate skeletal phenotypes that resemble human skeletal diseases. This review will also discuss other attractive aspects of the teleost skeleton. This includes the capacity for lifelong tooth replacement and for the regeneration of dermal skeletal elements, such as scales and fin rays, which further increases the value of zebrafish and medaka models for skeletal research.
authors.affiliation: Ghent University, Ghent, Belgium.
authors.forename: P E
authors.initials: PE
authors.lastname: Witten
authors.affiliation: Harvard Medical School, Boston, MA, United States.
authors.forename: M P
authors.initials: MP
authors.lastname: Harris
authors.affiliation: Ghent University, Ghent, Belgium.
authors.forename: A
authors.initials: A
authors.lastname: Huysseune
authors.affiliation: National University of Singapore, Singapore, Singapore.
authors.forename: C
authors.initials: C
authors.lastname: Winkler
mesh_terms: D000818:Animals; D030801:Animals, Genetically Modified / Q000235:genetics / Q000254:growth & development; D001846:Bone Development / Q000235:genetics; D001848:Bone Diseases, Developmental / Q000235:genetics* / Q000503:physiopathology; D064113:CRISPR-Cas Systems / Q000235:genetics; D004195:Disease Models, Animal; D006801:Humans; D008967:Molecular Biology / Q000379:methods*; D009990:Oryzias / Q000235:genetics* / Q000254:growth & development; D012038:Regeneration / Q000235:genetics; D000069896:Transcription Activator-Like Effector Nucleases / Q000235:genetics; D015027:Zebrafish / Q000235:genetics* / Q000254:growth & development
chemical_list: D000069896:Transcription Activator-Like Effector Nucleases
keywords: Bone; Cartilage; Chondrocytes; Genome editing; Medaka; Osteoblasts; Osteoclasts; Osteoporosis; Teeth; Zebrafish