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Biomedicines
Special Issues
Zebrafish Models for Development and Disease
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Zebrafish Models for Development and Disease

A special issue of Biomedicines (ISSN 2227-9059). This special issue belongs to the section "Model Systems in Research and Development".

Deadline for manuscript submissions: closed (30 June 2019) | Viewed by 42443

Special Issue Editors

Prof. Dr. James A. Marrs

E-Mail Website
Guest Editor
Department of Biology, Indiana University-Purdue University Indianapolis, Indianapolis, IN, USA
Interests: zebrafish; fetal alcohol spectrum disorder; gastrulation; congenital heart defects; eye defects; cadherin; tight junction; adherens junction
Special Issues, Collections and Topics in MDPI journals
Dr. Swapnalee Sarmah

E-Mail Website
Guest Editor
Department of Biology, Indiana University-Purdue University Indianapolis, Indianapolis, IN, USA
Interests: fetal alcohol spectrum disorder; congenital heart defects; zebrafish; craniofacial morphogenesis
Special Issues, Collections and Topics in MDPI journals

Special Issue Information

Dear Colleagues,

The zebrafish became an important model organism to study normal development, particularly after large scale genetic screening was used to identify genes that control developmental mechanisms. Subsequently, a large variety of creative approaches were used to develop disease models that are used to study developmental disorders, cancer, heart disease, diabetes, and many other conditions. The advent of next-generation DNA sequencing techniques expanded the utility of the zebrafish model, allowing analysis of genetic and epigenetic mechanisms of development and disease. Additionally, the small size, imaging capabilities and reporter gene expression in the zebrafish permits high throughput toxicology evaluation and drug screening, increasing the capability of this model. This call for papers invites contributions of original research and reviews for this Special Issue of Biomedicines entitled “Zebrafish Models in Development and Disease”. The Special Issue will explore the diverse capabilities of the zebrafish model that can be applied to study a growing list of biological and preclinical research problems.

Scope:

  • Basic research using the zebrafish model to understand diseases and mechanisms.
  • Basic research using the zebrafish to model disease treatment strategies.
  • Basic research using the zebrafish to evaluate potential teratogens.
  • Basic research using the zebrafish for drug screening.

Review articles on describing any of the above topics using the zebrafish model organism.

Prof. James A. Marrs
Dr. Swapnalee Sarmah
Guest Editors

Manuscript Submission Information

Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. Biomedicines is an international peer-reviewed open access monthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. The Article Processing Charge (APC) for publication in this open access journal is 2600 CHF (Swiss Francs). Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Keywords

  • Disease model
  • drug screening model
  • teratogen screening
  • toxicological screening
  • developmental biology

Related Special Issues

Published Papers (5 papers)

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Research

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15 pages, 3566 KiB  
Article
The Glycogen Synthase Kinase-3β Inhibitor LSN 2105786 Promotes Zebrafish Fin Regeneration
by Swapnalee Sarmah, Courtney Curtis, Jennifer Mahin, Mark Farrell, Thomas A. Engler, Manuel V. Sanchez-Felix, Masahiko Sato, Yanfai Linda Ma, Shaoyou Chu and James A. Marrs
Biomedicines 2019, 7(2), 30; https://doi.org/10.3390/biomedicines7020030 - 19 Apr 2019
Cited by 2 | Viewed by 4251
Abstract
The Wnt pathway has been shown to regulate bone homeostasis and to influence some bone disease states. We utilized a zebrafish model system to study the effects of a synthetic, orally bioavailable glycogen synthase kinase-3β (GSK3β) inhibitor LSN 2105786, which activates Wnt signaling [...] Read more.
The Wnt pathway has been shown to regulate bone homeostasis and to influence some bone disease states. We utilized a zebrafish model system to study the effects of a synthetic, orally bioavailable glycogen synthase kinase-3β (GSK3β) inhibitor LSN 2105786, which activates Wnt signaling during bone healing and embryogenesis. GSK3β inhibitor treatment was used to phenocopy GSK3β morpholino oligonucleotide (MO) knockdown in zebrafish embryos. Human and zebrafish synthetic mRNA injection were similarly effective at rescue of GSK3β MO knockdown. During caudal fin regeneration, bony rays are the first structure to differentiate in zebrafish fins, providing a useful model to study bone healing. Caudal fin regeneration experiments were conducted using various concentrations of a GSK3β inhibitor, examining duration and concentration dependence on regenerative outgrowth. Experiments revealed continuous low concentration (4–5 nM) treatment to be more effective at increasing regeneration than intermittent dosing. Higher concentrations inhibited fin growth, perhaps by excessive stimulation of differentiation programs. Increased Wnt responsive gene expression and differentiation were observed in response to GSK3b inhibitor treatment. Activating Wnt signaling also increased cell proliferation and osteoblast differentiation in fin regenerates. Together, these data indicate that bone healing in zebrafish fin regeneration was improved by activating Wnt signaling using GSK3b inhibitor treatment. In addition, caudal fin regeneration is useful to evaluate dose-dependent pharmacological efficacy in bone healing, various dosing regimens and possible toxicological effects of compounds. Full article
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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19 pages, 3146 KiB  
Article
A High-Throughput Assay for Congenital and Age-Related Eye Diseases in Zebrafish
by Lindy K. Brastrom, C. Anthony Scott, Deborah V. Dawson and Diane C. Slusarski
Biomedicines 2019, 7(2), 28; https://doi.org/10.3390/biomedicines7020028 - 11 Apr 2019
Cited by 13 | Viewed by 4505
Abstract
Debilitating visual impairment caused by cataracts or microphthalmia is estimated to affect roughly 20 million people in the United States alone. According to the National Eye Institute, by 2050 that number is expected to more than double to roughly 50 million. The identification [...] Read more.
Debilitating visual impairment caused by cataracts or microphthalmia is estimated to affect roughly 20 million people in the United States alone. According to the National Eye Institute, by 2050 that number is expected to more than double to roughly 50 million. The identification of candidate disease-causing alleles for cataracts and microphthalmia has been accelerated with advanced sequencing technologies creating a need for verification of the pathophysiology of these genes. Zebrafish pose many advantages as a high-throughput model for human eye disease. By 5 days post-fertilization, zebrafish have quantifiable behavioral responses to visual stimuli. Their small size, many progeny, and external fertilization allows for rapid screening for vision defects. We have adapted the OptoMotor Response to assay visual impairment in zebrafish models of cataracts and microphthalmia. This research demonstrates an inexpensive, high-throughput method for analyzing candidate genes involved in visual impairment. Full article
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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Review

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8 pages, 480 KiB  
Review
Zebrafish: A Powerful Model for Understanding the Functional Relevance of Noncoding Region Mutations in Human Genetic Diseases
by Anita Mann and Shipra Bhatia
Biomedicines 2019, 7(3), 71; https://doi.org/10.3390/biomedicines7030071 - 16 Sep 2019
Cited by 5 | Viewed by 3000
Abstract
Determining aetiology of genetic disorders caused by damaging mutations in protein-coding genes is well established. However, understanding how mutations in the vast stretches of the noncoding genome contribute to genetic abnormalities remains a huge challenge. Cis-regulatory elements (CREs) or enhancers are an important [...] Read more.
Determining aetiology of genetic disorders caused by damaging mutations in protein-coding genes is well established. However, understanding how mutations in the vast stretches of the noncoding genome contribute to genetic abnormalities remains a huge challenge. Cis-regulatory elements (CREs) or enhancers are an important class of noncoding elements. CREs function as the primary determinants of precise spatial and temporal regulation of their target genes during development by serving as docking sites for tissue-specific transcription factors. Although a large number of potential disease-associated CRE mutations are being identified in patients, lack of robust methods for mechanistically linking these mutations to disease phenotype is currently hampering the understanding of their roles in disease aetiology. Here, we have described the various systems available for testing the CRE potential of stretches of noncoding regions harbouring mutations implicated in human disease. We highlight advances in the field leading to the establishment of zebrafish as a powerful system for robust and cost-effective functional assays of CRE activity, enabling rapid identification of causal variants in regulatory regions and the validation of their role in disruption of appropriate gene expression. Full article
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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16 pages, 1203 KiB  
Review
Zebrafish Larvae as a Behavioral Model in Neuropharmacology
by Ram Manohar Basnet, Daniela Zizioli, Somrat Taweedet, Dario Finazzi and Maurizio Memo
Biomedicines 2019, 7(1), 23; https://doi.org/10.3390/biomedicines7010023 - 26 Mar 2019
Cited by 202 | Viewed by 20428
Abstract
Zebrafish larvae show a clear and distinct pattern of swimming in response to light and dark conditions, following the development of a swim bladder at 4 days post fertilization. This swimming behavior is increasingly employed in the screening of neuroactive drugs. The recent [...] Read more.
Zebrafish larvae show a clear and distinct pattern of swimming in response to light and dark conditions, following the development of a swim bladder at 4 days post fertilization. This swimming behavior is increasingly employed in the screening of neuroactive drugs. The recent emergence of high-throughput techniques for the automatic tracking of zebrafish larvae has further allowed an objective and efficient way of finding subtle behavioral changes that could go unnoticed during manual observations. This review highlights the use of zebrafish larvae as a high-throughput behavioral model for the screening of neuroactive compounds. We describe, in brief, the behavior repertoire of zebrafish larvae. Then, we focus on the utilization of light-dark locomotion test in identifying and screening of neuroactive compounds. Full article
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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16 pages, 606 KiB  
Review
On Zebrafish Disease Models and Matters of the Heart
by Panagiota Giardoglou and Dimitris Beis
Biomedicines 2019, 7(1), 15; https://doi.org/10.3390/biomedicines7010015 - 28 Feb 2019
Cited by 45 | Viewed by 8139
Abstract
Coronary artery disease (CAD) is the leading form of cardiovascular disease (CVD), which is the primary cause of mortality worldwide. It is a complex disease with genetic and environmental risk factor contributions. Reports in human and mammalian models elucidate age-associated changes in cardiac [...] Read more.
Coronary artery disease (CAD) is the leading form of cardiovascular disease (CVD), which is the primary cause of mortality worldwide. It is a complex disease with genetic and environmental risk factor contributions. Reports in human and mammalian models elucidate age-associated changes in cardiac function. The diverse mechanisms involved in cardiac diseases remain at the center of the research interest to identify novel strategies for prevention and therapy. Zebrafish (Danio rerio) have emerged as a valuable vertebrate model to study cardiovascular development over the last few decades. The facile genetic manipulation via forward and reverse genetic approaches combined with noninvasive, high-resolution imaging and phenotype-based screening has provided new insights to molecular pathways that orchestrate cardiac development. Zebrafish can recapitulate human cardiac pathophysiology due to gene and regulatory pathways conservation, similar heart rate and cardiac morphology and function. Thus, generations of zebrafish models utilize the functional analysis of genes involved in CAD, which are derived from large-scale human population analysis. Here, we highlight recent studies conducted on cardiovascular research focusing on the benefits of the combination of genome-wide association studies (GWAS) with functional genomic analysis in zebrafish. We further summarize the knowledge obtained from zebrafish studies that have demonstrated the architecture of the fundamental mechanisms underlying heart development, homeostasis and regeneration at the cellular and molecular levels. Full article
(This article belongs to the Special Issue Zebrafish Models for Development and Disease)
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