Despite the small size of the zebrafish, many traditional techniques for skeletal phenotyping, such as x-ray and microCT imaging and histological approaches, can be applied using the appropriate equipment and custom protocols. In addition, transgenic lines expressing fluorescent proteins under bone cell- or pathway- specific promoters enable in vivo imaging of differentiation and signaling at the cellular level. In the last decades, the use of both forward and new reverse genetics techniques has resulted in the generation of many mutant lines carrying skeletal phenotypes associated with human diseases. Furthermore, zebrafish share similar skeletal cells and ossification types with mammals. The zebrafish has been used as an efficient alternative vertebrate model for the study of human skeletal diseases, thanks to its easy genetic manipulation, high fecundity, external fertilization, transparency of rapidly developing embryos, and low maintenance cost. 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. 2Department of Biomolecular Medicine, Center of Medical Genetics, Ghent University-University Hospital, Ghent, BelgiumĪnimal models are essential tools for addressing fundamental scientific questions about skeletal diseases and for the development of new therapeutic approaches.1Biochemistry Unit, Department of Molecular Medicine, University of Pavia, Pavia, Italy.
Coucke 2, Andy Willaert 2 ‡ and Antonella Forlino 1 * ‡ Francesca Tonelli 1 †, Jan Willem Bek 2 †, Roberta Besio 1 †, Adelbert De Clercq 2 †, Laura Leoni 1, Phil Salmon 3, Paul J.
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