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      Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques

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          Abstract

          For the past few years there has been an exponential increase in the use of animal models to confirm the pathogenicity of candidate disease-causing genetic variants found in patients. One such animal model is the zebrafish. Despite being a non-mammalian animal, the zebrafish model has proven its potential in recapitulating the phenotypes of many different human genetic disorders. This review will focus on recent advances in the modeling of neurodevelopmental disorders in zebrafish, covering aspects from early brain development to techniques used for modulating gene expression, as well as how to best characterize the resulting phenotypes. We also review other existing models of neurodevelopmental disorders, and the current efforts in developing and testing compounds with potential therapeutic value.

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          The zebrafish reference genome sequence and its relationship to the human genome.

          Kerstin Howe, Matthew D. Clark, Carlos F Torroja … (2013)
          Zebrafish have become a popular organism for the study of vertebrate gene function. The virtually transparent embryos of this species, and the ability to accelerate genetic studies by gene knockdown or overexpression, have led to the widespread use of zebrafish in the detailed investigation of vertebrate gene function and increasingly, the study of human genetic disease. However, for effective modelling of human genetic disease it is important to understand the extent to which zebrafish genes and gene structures are related to orthologous human genes. To examine this, we generated a high-quality sequence assembly of the zebrafish genome, made up of an overlapping set of completely sequenced large-insert clones that were ordered and oriented using a high-resolution high-density meiotic map. Detailed automatic and manual annotation provides evidence of more than 26,000 protein-coding genes, the largest gene set of any vertebrate so far sequenced. Comparison to the human reference genome shows that approximately 70% of human genes have at least one obvious zebrafish orthologue. In addition, the high quality of this genome assembly provides a clearer understanding of key genomic features such as a unique repeat content, a scarcity of pseudogenes, an enrichment of zebrafish-specific genes on chromosome 4 and chromosomal regions that influence sex determination.
          Article 0 comments Cited 1443 times – based on 0 reviews     Review now
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            The Tol2kit: a multisite gateway-based construction kit for Tol2 transposon transgenesis constructs.

            Kristen Kwan, Esther Fujimoto, Clemens Grabher … (2007)
            Transgenesis is an important tool for assessing gene function. In zebrafish, transgenesis has suffered from three problems: the labor of building complex expression constructs using conventional subcloning; low transgenesis efficiency, leading to mosaicism in transient transgenics and infrequent germline incorporation; and difficulty in identifying germline integrations unless using a fluorescent marker transgene. The Tol2kit system uses site-specific recombination-based cloning (multisite Gateway technology) to allow quick, modular assembly of [promoter]-[coding sequence]-[3' tag] constructs in a Tol2 transposon backbone. It includes a destination vector with a cmlc2:EGFP (enhanced green fluorescent protein) transgenesis marker and a variety of widely useful entry clones, including hsp70 and beta-actin promoters; cytoplasmic, nuclear, and membrane-localized fluorescent proteins; and internal ribosome entry sequence-driven EGFP cassettes for bicistronic expression. The Tol2kit greatly facilitates zebrafish transgenesis, simplifies the sharing of clones, and enables large-scale projects testing the functions of libraries of regulatory or coding sequences. Copyright 2007 Wiley-Liss, Inc.
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              Fluorescent indicators for Ca2+ based on green fluorescent proteins and calmodulin.

              A Miyawaki, J Llopis, R. Heim … (1997)
              Important Ca2+ signals in the cytosol and organelles are often extremely localized and hard to measure. To overcome this problem we have constructed new fluorescent indicators for Ca2+ that are genetically encoded without cofactors and are targetable to specific intracellular locations. We have dubbed these fluorescent indicators 'cameleons'. They consist of tandem fusions of a blue- or cyan-emitting mutant of the green fluorescent protein (GFP), calmodulin, the calmodulin-binding peptide M13, and an enhanced green- or yellow-emitting GFP. Binding of Ca2+ makes calmodulin wrap around the M13 domain, increasing the fluorescence resonance energy transfer (FRET) between the flanking GFPs. Calmodulin mutations can tune the Ca2+ affinities to measure free Ca2+ concentrations in the range 10(-8) to 10(-2) M. We have visualized free Ca2+ dynamics in the cytosol, nucleus and endoplasmic reticulum of single HeLa cells transfected with complementary DNAs encoding chimaeras bearing appropriate localization signals. Ca2+ concentration in the endoplasmic reticulum of individual cells ranged from 60 to 400 microM at rest, and 1 to 50 microM after Ca2+ mobilization. FRET is also an indicator of the reversible intermolecular association of cyan-GFP-labelled calmodulin with yellow-GFP-labelled M13. Thus FRET between GFP mutants can monitor localized Ca2+ signals and protein heterodimerization in individual live cells.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Int J Mol Sci
                Journal ID (iso-abbrev): Int J Mol Sci
                Journal ID (publisher-id): ijms
                Title: International Journal of Molecular Sciences
                Publisher: MDPI
                ISSN (Electronic): 1422-0067
                Publication date (Electronic): 14 March 2019
                Publication date Collection: March 2019
                Volume: 20
                Issue: 6
                Electronic Location Identifier: 1296
                Affiliations
                [1 ]Department of Molecular Medicine and Surgery, Center for Molecular Medicine, Karolinska Institutet, 171 76 Stockholm, Sweden; raquel.vaz@ 123456ki.se
                [2 ]Laboratory of Molecular and Cellular Cardiology, Department of Clinical Biochemistry and Pharmacology, Odense University Hospital, 5000 Odense, Denmark; hofmeister@ 123456health.sdu.dk
                [3 ]Novo Nordisk Foundation for Stem Cell Biology (Danstem), University of Copenhagen, 2200 Copenhagen, Denmark
                [4 ]Department of Molecular Medicine and Surgery, Center for Molecular Medicine and Clinical Genetics, Karolinska University Laboratory, Karolinska University Hospital, 171 76 Stockholm, Sweden
                Author notes
                [* ]Correspondence: anna.lindstrand@ 123456ki.se ; Tel.: +46-8-517-765-38
                Author information
                https://orcid.org/0000-0001-7518-1801
                https://orcid.org/0000-0002-6306-9262
                https://orcid.org/0000-0003-0806-5602
                Article
                Publisher ID: ijms-20-01296
                DOI: 10.3390/ijms20061296
                PMC ID: 6471844
                PubMed ID: 30875831
                SO-VID: d5a00030-9eb1-4eb3-858b-a97fed57a138
                Copyright © © 2019 by the authors.
                License:

                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 ( http://creativecommons.org/licenses/by/4.0/).

                History
                Date received : 29 January 2019
                Date accepted : 11 March 2019
                Categories
                Subject: Review

                ScienceOpen disciplines: Molecular biology
                Keywords: zebrafish,neurodevelopmental disorders,disease models,functional assays
                Data availability:
                ScienceOpen disciplines: Molecular biology
                Keywords: zebrafish, neurodevelopmental disorders, disease models, functional assays

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