2020
DOI: 10.3389/fendo.2020.00489
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Zebrafish: A Resourceful Vertebrate Model to Investigate Skeletal Disorders

Abstract: Animal models are essential tools for addressing fundamental scientific questions about skeletal diseases and for the development of new therapeutic approaches. Traditionally, mice have been the most common model organism in biomedical research, but their use is hampered by several limitations including complex generation, demanding investigation of early developmental stages, regulatory restrictions on breeding, and high maintenance cost. The zebrafish has been used as an efficient alternative vertebrate mode… Show more

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Cited by 80 publications
(65 citation statements)
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“…The recently established CRISPR-Cas9 gene editing additionally increased the value of this model. Zebrafish are also particularly suitable for small-molecule drug application, which, in skeletal field, takes enormous advantage of tail regeneration [ 47 ].…”
Section: Experimental Approachesmentioning
confidence: 99%
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“…The recently established CRISPR-Cas9 gene editing additionally increased the value of this model. Zebrafish are also particularly suitable for small-molecule drug application, which, in skeletal field, takes enormous advantage of tail regeneration [ 47 ].…”
Section: Experimental Approachesmentioning
confidence: 99%
“…Zebrafish is not only a fascinating example of vertebrate regeneration, but also represents a well-known vertebrate model for disease and has already contributed to several examples of successful phenotype-based drug discovery, providing a tool with many different applications in biomedical research, especially in the field of bone biology. Indeed, zebrafish skeleton conserves the general basis of development, gene expression and cell types that are found in mammals [ 47 ]. In this scenario, the remarkably fast regeneration of zebrafish caudal fin provides an excellent tool to easy monitor bone formation over time in healthy and pathological conditions [ 47 ].…”
Section: Regeneration Models As a Tool For Biomedical Researchmentioning
confidence: 99%
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“…Zebrafish have several advantages over mice, making experiments quicker and less expensive: they are highly fecund, laying up to 300 eggs a week; phenotypes may be evident at the larval stage (skeletal elements develop by four days); embryos develop externally, enabling genetic manipulation at the single cell stage. In addition, larvae are translucent, allowing dynamic visualization of skeletal cell behavior, for which several transgenic reporter lines are available ( 77 , 78 ). Embryonic lethality is rare with fish able to survive despite mutations leading to a complete absence of bone tissue, as they are supported by water as they swim, which limits loading of malformed skeletal elements ( 79 ).…”
Section: Functional Genomics: In Vivo Studiesmentioning
confidence: 99%
“…Although, endochondral ossification is not as frequent as in mammals and additionally, intramembranous and perichondral ossification is present in zebrafish [ 79 ]. Investigation of TNAP function in zebrafish is still fragmented but may open up the possibility to investigate more basic developmental aspects due to its biological features, like extracorporeal fertilization, optical transparency, and numerous transgenic fluorescent reporter lines [ 77 , 80 ]. Genetic analyses of alpl gene evolution in vertebrates and their expression in zebrafish clarified that non-mammalian species possess a number of additional alpl genes due to genome duplication events [ 8 ].…”
Section: The Molecular Role Of Tnap In Bone Mineralizationmentioning
confidence: 99%