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Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies

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 st...

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Autores principales: Santos, João M. A., Laizé, Vincent, Gavaia, Paulo J., Conceição, Natércia, Cancela, M. Leonor
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9967340/
https://www.ncbi.nlm.nih.gov/pubmed/36834795
http://dx.doi.org/10.3390/ijms24043366
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author Santos, João M. A.
Laizé, Vincent
Gavaia, Paulo J.
Conceição, Natércia
Cancela, M. Leonor
author_facet Santos, João M. A.
Laizé, Vincent
Gavaia, Paulo J.
Conceição, Natércia
Cancela, M. Leonor
author_sort Santos, João M. A.
collection PubMed
description 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 (Danio rerio)—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.
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spelling pubmed-99673402023-02-26 Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies Santos, João M. A. Laizé, Vincent Gavaia, Paulo J. Conceição, Natércia Cancela, M. Leonor Int J Mol Sci Review 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 (Danio rerio)—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. MDPI 2023-02-08 /pmc/articles/PMC9967340/ /pubmed/36834795 http://dx.doi.org/10.3390/ijms24043366 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Review
Santos, João M. A.
Laizé, Vincent
Gavaia, Paulo J.
Conceição, Natércia
Cancela, M. Leonor
Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title_full Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title_fullStr Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title_full_unstemmed Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title_short Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies
title_sort zebrafish models to study ectopic calcification and calcium-associated pathologies
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9967340/
https://www.ncbi.nlm.nih.gov/pubmed/36834795
http://dx.doi.org/10.3390/ijms24043366
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