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Published on: October 18, 2018
Zebrafish Models for Human Skeletal Disorders
Manuel Marí-Beffa1,2, Ana B Mesa-Román1, Ivan Duran1,2
1Department of Cell Biology, Genetics and Physiology, Faculty of Sciences, University of Málaga, IBIMA, Málaga, Spain.
Abstract:
In 2019, the Nosology Committee of the International Skeletal Dysplasia Society provided an updated version of the Nosology and Classification of Genetic Skeletal Disorders. This is a reference list of recognized diseases in humans and their causal genes published to help clinician diagnosis and scientific research advances. Complementary to mammalian models, zebrafish has emerged as an interesting species to evaluate chemical treatments against these human skeletal disorders. Due to its versatility and the low cost of experiments, more than 80 models are currently available. In this article, we review the state-of-art of this "aquarium to bedside" approach describing the models according to the list provided by the Nosology Committee. With this, we intend to stimulate research in the appropriate direction to efficiently meet the actual needs of clinicians under the scope of the Nosology Committee.
Insights
Zebrafish models offer a versatile platform for studying human skeletal dysplasias, aiding in the development of new chemical treatments. This review aligns zebrafish research with the International Skeletal Dysplasia Society
Area of Science:
- Genetics
- Developmental Biology
- Pharmacology
Background:
- The International Skeletal Dysplasia Society updated its classification of genetic skeletal disorders in 2019.
- Understanding the genetic basis of these disorders is crucial for diagnosis and research.
- Zebrafish offer a valuable model system for studying human skeletal diseases.
Purpose of the Study:
- To review the current state of zebrafish models for genetic skeletal disorders.
- To align zebrafish research with the 2019 Nosology and Classification of Genetic Skeletal Disorders.
- To encourage research that bridges the gap between zebrafish studies and clinical applications.
Main Methods:
- Review of existing literature on zebrafish models for skeletal dysplasias.
- Categorization of zebrafish models based on the 2019 Nosology Committee's classification.
- Analysis of the 'aquarium to bedside' approach in this field.
Main Results:
- Over 80 zebrafish models for skeletal disorders are currently available.
- Zebrafish models provide a cost-effective and versatile platform for research.
- The review systematically presents these models according to the established nosology.
Conclusions:
- Zebrafish research is a promising avenue for developing treatments for human skeletal dysplasias.
- Aligning research with clinical nosology is essential for translational success.
- Further research is needed to fully leverage zebrafish models for clinical benefit.

