Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies

• Published in: International journal of molecular sciences Vol. 24; no. 4; p. 3366
• Main Authors: Santos, João M A, Laizé, Vincent, Gavaia, Paulo J, Conceição, Natércia, Cancela, M Leonor
• Format: Journal Article
• Language: English
• Published: Switzerland MDPI AG 01.02.2023; MDPI
• Subjects: Age; Animals; Bone growth; Calcification; Calcification (ectopic); Calcification, Physiologic; Calcinosis - metabolism; Calcium; Calcium - metabolism; Calcium ions; Calcium, Dietary - metabolism; Cartilage; Danio rerio; disease modeling; DNA damage; ectopic calcification; Extracellular matrix; Extracellular Matrix - metabolism; Genetic disorders; Genetic engineering; Genotype & phenotype; Heart; Homeostasis; Humans; Kidney diseases; Medical research; Medicine, Experimental; Metabolism; Metastasis; Mice; Mineralization; Mutants; Mutation; Osteogenesis; Pathophysiology; Phenotypes; Proteins; Review; Soft tissues; Zebrafish; Zebrafish - genetics; mineralization; calcium; zebrafish; ectopic calcification; disease modeling
• ISSN: 1422-0067; 1661-6596; 1422-0067
• DOI: 10.3390/ijms24043366

Ectopic calcification refers to the pathological accumulation of calcium ions in soft tissues and is often the result of a dysregulated action or disrupted function of proteins involved in extracellular matrix mineralization. While the mouse has traditionally been the go-to model organism for the study of pathologies associated with abnormal calcium deposition, many mouse mutants often have exacerbated phenotypes and die prematurely, limiting the understanding of the disease and the development of effective therapies. Since the mechanisms underlying ectopic calcification share some analogy with those of bone formation, the zebrafish ( )-a well-established model for studying osteogenesis and mineralogenesis-has recently gained momentum as a model to study ectopic calcification disorders. In this review, we outline the mechanisms of ectopic mineralization in zebrafish, provide insights into zebrafish mutants that share phenotypic similarities with human pathological mineralization disorders, list the compounds capable of rescuing mutant phenotypes, and describe current methods to induce and characterize ectopic calcification in zebrafish.