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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| Veröffentlicht in: | International journal of molecular sciences 2023-02, Vol.24 (4), p.3366 |
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| creator | Santos, João M A Laizé, Vincent Gavaia, Paulo J Conceição, Natércia Cancela, M Leonor |
| 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 (
)-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. |
| doi_str_mv | 10.3390/ijms24043366 |
| format | Article |
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)-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.</description><identifier>ISSN: 1422-0067</identifier><identifier>ISSN: 1661-6596</identifier><identifier>EISSN: 1422-0067</identifier><identifier>DOI: 10.3390/ijms24043366</identifier><identifier>PMID: 36834795</identifier><language>eng</language><publisher>Switzerland: MDPI AG</publisher><subject>Age ; Animals ; Bone growth ; Calcification ; Calcification (ectopic) ; Calcification, Physiologic ; Calcinosis - metabolism ; Calcium ; Calcium - metabolism ; Calcium ions ; Calcium, Dietary - metabolism ; Cartilage ; Danio rerio ; DNA damage ; 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</subject><ispartof>International journal of molecular sciences, 2023-02, Vol.24 (4), p.3366</ispartof><rights>COPYRIGHT 2023 MDPI AG</rights><rights>2023 by the authors. 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/). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2023 by the authors. 2023</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c507t-53d07356c00829bc70372f036b7c394d56fda71c2d92fcc9384a79b119ee57683</citedby><cites>FETCH-LOGICAL-c507t-53d07356c00829bc70372f036b7c394d56fda71c2d92fcc9384a79b119ee57683</cites><orcidid>0000-0003-3114-6662 ; 0000-0002-5057-0912 ; 0000-0002-9582-1957 ; 0000-0002-1218-368X ; 0000-0001-9565-9198</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9967340/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9967340/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,881,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36834795$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Santos, João M A</creatorcontrib><creatorcontrib>Laizé, Vincent</creatorcontrib><creatorcontrib>Gavaia, Paulo J</creatorcontrib><creatorcontrib>Conceição, Natércia</creatorcontrib><creatorcontrib>Cancela, M Leonor</creatorcontrib><title>Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies</title><title>International journal of molecular sciences</title><addtitle>Int J Mol Sci</addtitle><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 (
)-a well-established model for studying osteogenesis and mineralogenesis-has recently gained momentum as a model to study ectopic calcification disorders. 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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.</abstract><cop>Switzerland</cop><pub>MDPI AG</pub><pmid>36834795</pmid><doi>10.3390/ijms24043366</doi><orcidid>https://orcid.org/0000-0003-3114-6662</orcidid><orcidid>https://orcid.org/0000-0002-5057-0912</orcidid><orcidid>https://orcid.org/0000-0002-9582-1957</orcidid><orcidid>https://orcid.org/0000-0002-1218-368X</orcidid><orcidid>https://orcid.org/0000-0001-9565-9198</orcidid><oa>free_for_read</oa></addata></record> |
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| source | MDPI - Multidisciplinary Digital Publishing Institute; MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central |
| subjects | Age Animals Bone growth Calcification Calcification (ectopic) Calcification, Physiologic Calcinosis - metabolism Calcium Calcium - metabolism Calcium ions Calcium, Dietary - metabolism Cartilage Danio rerio DNA damage 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 |
| title | Zebrafish Models to Study Ectopic Calcification and Calcium-Associated Pathologies |
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