Zebrafish Developmental Models of Skeletal Diseases

• Published in: Current topics in developmental biology Vol. 124; pp. 81 - 124
• Main Authors: Luderman, Lauryn N, Unlu, Gokhan, Knapik, Ela W
• Format: Journal Article
• Language: English
• Published: United States 2017
• Subjects: Animals; Bone Diseases - pathology; Cartilage - embryology; Disease Models, Animal; Extracellular Matrix - metabolism; Humans; Macromolecular Substances - metabolism; Zebrafish - embryology; Development; Cartilage; Skeletal syndromes; Zebrafish; Bone
• ISSN: 1557-8933
• DOI: 10.1016/bs.ctdb.2016.11.004

The zebrafish skeleton shares many similarities with human and other vertebrate skeletons. Over the past years, work in zebrafish has provided an extensive understanding of the basic developmental mechanisms and cellular pathways directing skeletal development and homeostasis. This review will focus on the cell biology of cartilage and bone and how the basic cellular processes within chondrocytes and osteocytes function to assemble the structural frame of a vertebrate body. We will discuss fundamental functions of skeletal cells in production and secretion of extracellular matrix and cellular activities leading to differentiation of progenitors to mature cells that make up the skeleton. We highlight important examples where findings in zebrafish provided direction for the search for genes causing human skeletal defects and also how zebrafish research has proven important for validating candidate human disease genes. The work we cover here illustrates utility of zebrafish in unraveling molecular mechanisms of cellular functions necessary to form and maintain a healthy skeleton.