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The identification of genes with unique and essential functions in the development of the zebrafish, Danio rerio

P Haffter, M Granato, M Brand, M C Mullins, M Hammerschmidt, D A Kane, J Odenthal, F J van Eeden, Y J Jiang, C P Heisenberg, R N Kelsh, M Furutani-Seiki (+5 others)
1996 Development  
In a large-scale screen, we isolated mutants displaying a specific visible phenotype in embryos or early larvae of the zebrafish, Danio rerio.  ...  F3 progeny were scored at the second, third and sixth day of development, using a stereomicroscope. In a subsequent screen, fixed embryos were analyzed for correct retinotectal projection.  ...  This work was a collaborative effort in which a large number of people participated during all or part of the three years of raising the families, screening and evaluation of the mutants.  ... 
pmid:9007226 fatcat:sn2a4ifhhveyblqzgshvmrtrsa

Zebrafish development and regeneration: new tools for biomedical research

Sebastiaan A. Brittijn, Suzanne J. Duivesteijn, Mounia Belmamoune, Laura F.M. Bertens, Wilbert Bitter, Joost D. Debruijn, Danielle L. Champagne, Edwin Cuppen, Gert Flik, Christina M. Vandenbroucke-Grauls, Richard A.J. Janssen, Ilse M.L. de Jong (+15 others)
2009 International Journal of Developmental Biology  
The assays use live zebrafish embryos and larvae as well as adult fish undergoing caudal fin regeneration.  ...  Our assays involve: (i) systemic or local administration of a test compound or drug to zebrafish in vivo; (ii) the subsequent detection or "readout" of a defined phenotypic change.  ...  We would like to thank Ben Nelemans for help with image editing and Edwin Heida for drawing Figures 1 and 9.  ... 
doi:10.1387/ijdb.082615sb pmid:19557689 fatcat:bsapoolrafd4bm4bd3dic7kr6m

The zebrafish:scalable in vivo modeling for systems biology

Rahul C. Deo, Calum A. MacRae
2010 Wiley Interdisciplinary Reviews: Systems Biology and Medicine  
The zebrafish offers a scalable vertebrate model for many areas of biologic investigation.  ...  We highlight recent work demonstrating the extent of this homology, and efforts to develop high-throughput phenotyping strategies suited to genetic or chemical screening on a scale compatible with in vivo  ...  a chemical gradient and the resulting migration of leukocytes-a task ideally suited for study in the zebrafish embryo. 61 The authors made use of genetically encoded H 2 O 2 sensor, HyPer, which consists  ... 
doi:10.1002/wsbm.117 pmid:20882534 pmcid:PMC3251331 fatcat:pzqffof72nalrmclqdmo63yuwm

Genome resource banking of biomedically important laboratory animals

Yuksel Agca
2012 Theriogenology  
In that regard, appropriate genome cryobanks could provide essential biomaterials for both current and future research projects in the form of various cell types and tissues, including sperm, oocytes,  ...  For revitalization of the whole organism, cryopreserved germplasm in conjunction with assisted reproductive technologies (ART), offer a powerful approach for research model management, as well as assisting  ...  also for in vitro drug and toxicity screening [111, 112] .  ... 
doi:10.1016/j.theriogenology.2012.08.012 pmid:22981880 pmcid:PMC3939794 fatcat:jable2dyazevro2rc6wlaf6pa4

Vertebrate Segmentation: From Cyclic Gene Networks to Scoliosis

Olivier Pourquié
2011 Cell  
In zebrafish embryos, the role of Notch in the clock pacemaker has been ruled out by the following three observations: (1) anterior somites do form in Notch mutants; (2) when using the g-secretase inhibitor  ...  Disruption of the coupling, as in the Notch pathway mutants in zebrafish or using DAPT treatment, increases the period of somitogenesis, which leads to the formation of larger segments (Herrgen et al.  ... 
doi:10.1016/j.cell.2011.05.011 pmid:21620133 pmcid:PMC3164975 fatcat:d4i63xoczfca5eebcsdcugej4m

Zebrafish models for ectopic mineralization disorders: practical issues from morpholino design to post-injection observations

Mohammad Hosen
2013 Frontiers in Genetics  
In this paper, we will discuss all details about the aforementioned techniques; shared knowledge will be very useful for the future investigation of ZF models for ectopic mineralization disorders and to  ...  To characterize the function of early mineralization-related genes in ZF, these genes can be knocked down by injecting morpholinos into early stage embryos.  ...  However, the need for screening large number of F1 fish to find a lesion in a gene of interest (Wienholds et al., 2002) , the requirement of specialized equipment, and a significant investment in computational  ... 
doi:10.3389/fgene.2013.00074 pmid:23760765 pmcid:PMC3669896 fatcat:k6f74mephfhjdie3vt6o4xc7bm

Generation of Rab-based transgenic lines for in vivo studies of endosome biology in zebrafish

Brian S. Clark, Mark Winter, Andrew R. Cohen, Brian A. Link
2011 Developmental Dynamics  
In summary, we have established a toolset for in vivo analyses of endosome dynamics and functions.  ...  To facilitate in vivo investigation of specific Rab proteins, and endosome biology in general, we have generated transgenic zebrafish lines to mark and manipulate Rab proteins.  ...  Clare Buckley, Jon Clarke, and Brian Perkins for comments and critically reviewing drafts of this manuscript.  ... 
doi:10.1002/dvdy.22758 pmid:21976318 pmcid:PMC3197968 fatcat:fqsvm2vi25dethjonzep7vg5um

The progress of CRISPR/Cas9-mediated gene editing in generating mouse/zebrafish models of human skeletal diseases

Nan Wu, Bowen Liu, Huakang Du, Sen Zhao, Yaqi Li, Xi Cheng, Shengru Wang, Jiachen Lin, Junde Zhou, Guixing Qiu, Jianguo Zhang, Zhihong Wu
2019 Computational and Structural Biotechnology Journal  
Animal models including mouse and zebrafish have been extensively used in functional mechanism studies of causal genes and variants.  ...  The conventional approaches of generating mutant animal models include spontaneous mutagenesis, random integration, and targeted integration via mouse embryonic stem cells.  ...  A variant in ATP6V1H was screened from hundreds of zebrafish mutants and identified as pathogenic mutation in the visual system [108] [109] [110] [111] .  ... 
doi:10.1016/j.csbj.2019.06.006 pmid:31360334 pmcid:PMC6639410 fatcat:feags2fqc5d7fopgpcmbpubo6i

The UNC-45 Myosin Chaperone [chapter]

Chi F. Lee, Girish C. Melkani, Sanford I. Bernstein
2014 International Review of Cell and Molecular Biology  
This was demonstrated using a titin reporter and atomic force microscopy as discussed in section 4.1. Dedication We dedicate this article to the memory of Dr. Henry F.  ...  It likely aides in folding myosin during cellular differentiation and maintenance and protects myosin from denaturation during stress.  ...  Work in our laboratory regarding UNC-45 is supported by National Institutes of Health grant 5R01AR055958 to SIB.  ... 
doi:10.1016/b978-0-12-800177-6.00004-9 pmid:25376491 pmcid:PMC4225561 fatcat:4jgg6nhysnf57d2sxequvhsxaa

The zebrafish as a promising tool for modeling human brain disorders: A review based upon an IBNS Symposium

Soaleha Shams, Jason Rihel, Jose G. Ortiz, Robert Gerlai
2018 Neuroscience and Biobehavioral Reviews  
disorder in larval zebrafish, on screening natural products for compounds with psychoactive properties in adult zebrafish, and on the development of a zebrafish model of fetal alcohol spectrum disorders  ...  Also promising are screens for psychoactive drugs with effects on larval and adult zebrafish behavior.  ...  We wish to thank members of the Rihel, Ortiz, and Gerlai labs for critical discussion and comments. Zebrafish models of human brain disorders 48 Shams et al.  ... 
doi:10.1016/j.neubiorev.2017.09.002 pmid:28887224 fatcat:ymtp4p6mlbd3xmoxfaxl5lor5q

Optic cup morphogenesis requires neural crest-mediated basement membrane assembly

Chase D. Bryan, Macaulie A. Casey, Rebecca L. Pfeiffer, Bryan W. Jones, Kristen M. Kwan
2020 Development  
Here, using multidimensional imaging and computational analyses in zebrafish, we establish that cell movements in the developing optic cup require neural crest.  ...  Neural crest cells produce the extracellular matrix protein nidogen: impairing nidogen function disrupts eye development, and, strikingly, expression of nidogen in the absence of neural crest partially  ...  Thanks to members of the Kwan lab for useful discussions and critical reading of the manuscript.  ... 
doi:10.1242/dev.181420 pmid:31988185 pmcid:PMC7044464 fatcat:rhtiwb4boje2pjf35r6hn6hjne

A high-throughput chemically induced inflammation assay in zebrafish

Claudia A d'Alençon, Oscar A Peña, Christine Wittmann, Viviana E Gallardo, Rebecca A Jones, Felix Loosli, Urban Liebel, Clemens Grabher, Miguel L Allende
2010 BMC Biology  
We demonstrate the usefulness of this method for chemical and genetic screens to detect the effect of immunomodulatory compounds and mutations affecting the leukocyte response.  ...  This approach allows rapid screening of thousands of compounds or mutagenized zebrafish for effects on inflammation and enables the identification of novel players in the regulation of innate immunity  ...  Here we provide proof of principle for the identification of mutations using was mutant zebrafish as an example.  ... 
doi:10.1186/1741-7007-8-151 pmid:21176202 pmcid:PMC3022775 fatcat:mgnr6sikjnhyfjvrok6px7gxom

The study of psychiatric disease genes and drugs in zebrafish

Martin Haesemeyer, Alexander F Schier
2015 Current Opinion in Neurobiology  
Recent zebrafish research has uncovered potential developmental phenotypes for genes associated with psychiatric disorders, while drug screens have behaviorally characterized small molecules and identified  ...  Behavioral studies have led to promising models for endophenotypes of psychiatric diseases.  ...  Acknowledgments We thank Florian Merkle, Iris Odstrcil and Summer Thyme for critical comments on the manuscript and the Jane Coffin Childs Fund as well as the NIH for funding.  ... 
doi:10.1016/j.conb.2014.12.002 pmid:25523356 pmcid:PMC4294547 fatcat:pbeoadrzqvdmzd74vsgtza7v2e

Use of Zebrafish in Drug Discovery Toxicology

Steven Cassar, Isaac Adatto, Jennifer L Freeman, Joshua T. Gamse, Iñaki Iturria, Christian Lawrence, Arantza Muriana, Randall Peterson, Steven Van Cruchten, Leonard Zon
2019 Chemical Research in Toxicology  
The zebrafish model is a bridge between in vitro assays and mammalian in vivo studies. This model is powerful in its breadth of application and tractability for research.  ...  Unpredicted human safety events in clinical trials for new drugs are costly in terms of human health and money.  ...  ■ EMBRYO TOXICITY Zebrafish embryos are increasingly used for developmental toxicity screening of candidate drugs and chemicals ( Figure 3) .  ... 
doi:10.1021/acs.chemrestox.9b00335 pmid:31625720 pmcid:PMC7162671 fatcat:ghwwy4xt3jeg7ap4ase27fqj4a

CDC14A phosphatase is essential for hearing and male fertility in mouse and human

Ayesha Imtiaz, Inna A Belyantseva, Alisha J Beirl, Cristina Fenollar-Ferrer, Rasheeda Bashir, Ihtisham Bukhari, Amal Bouzid, Uzma Shaukat, Hela Azaiez, Kevin T Booth, Kimia Kahrizi, Hossein Najmabadi (+19 others)
2017 Human Molecular Genetics  
In mutant male mice, degeneration of seminiferous tubules and spermiation defects result in low sperm count, and abnormal sperm motility and morphology.  ...  We generated and analyzed mutations of zebrafish and mouse CDC14A, developed a computational structural model of human CDC14A protein and report four novel truncating and three missense alleles of CDC14A  ...  using Revert Aid tm Premium (Fermentas).  ... 
doi:10.1093/hmg/ddx440 pmid:29293958 pmcid:PMC6059191 fatcat:klzblu5mp5attne3g4sjdxvnnq
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