13:e1007000. The combination of these new tools for linking neurons to behavior with genetic approaches and analysis of mutants make zebrafish an ideal model to bridge the challenging gaps between genes, neurons, and behavior. Taking substantiated decisions in Zebrafish research. Detection of enteric neural crest cells (ENCCs) and enteric neurons in zebrafish, is the vital first step to study ENS development and defects herein. (2016). Larval zebrafish can be housed in 96-well plates, where they can be exposed to small molecule libraries for drug discovery. Nat. This reduces variability due to differences in food intake, but increases the manual labor and expertise required to perform this technique. “Enteric nervous system,” in Encyclopedia of Neuroscience, eds M. D. Binder, N. Hirokawa, and U. Windhorst (Berlin, Heidelberg: Springer Berlin Heidelberg), 1122–1125. Whole-organism clone tracing using single-cell sequencing. Research often utilizes patients’ cells or tissue samples, but to determine if a mutation in a specific gene can cause a patient’s symptoms, we often need experimental animal models. Stephanie Padilla, Scott Glaberman, in An Introduction to Interdisciplinary Toxicology, 2020. Development 123, 1–36. They also show that, zebrafish enteric glia resemble mammalian enteric glia based on their ultrastructural and general morphology, gene expression and location. (2013). PLoS Biol. In case of reduced Ret expression by MO, disruption of the Glutamine synthetase gene (gnl1) also resulted in increased occurrence of HSCR phenotypes. Sci. Their embryos develop very rapidly, hatching after about 50 h in controlled conditions, and their larvae begin to eat starting on day 5.Zebrafish are exotherms and are typically cultured at 28°C, though wild zebrafish have been shown to populate diverse habitats from 18°C to over 38°C. Genet. doi: 10.1053/j.gastro.2020.07.018, Spanjaard, B., Hu, B., Mitic, N., Olivares-Chauvet, P., Janjuha, S., Ninov, N., et al. Edward A. Burton, in Movement Disorders (Second Edition), 2015. Over the past decade, zebrafish have been gaining increasing attention in behavioral neuroscience due to a number of advantages over several vertebrate and invertebrate models. A second contribution to the ENS arises from sacral NCCs that seed the distal part of the GI tract with enteric ganglia. Development 140, 2835–2846. Comp. The Tol2-mediated Gal4-UAS method for gene and enhancer trapping in zebrafish. In the next sections, we discuss some of the approaches currently used to genetically modify or block, expression of specific ENS genes using zebrafish, in an attempt to provide insights into the genetics and mechanisms underlying HSCR development. Gastroenterology 155, 118–129.e6. Recently, a rather non-conventional way to label enteric neurons was developed by applying a local heat shock to induce a genetic switch of the fluorophore expression, in the zebrabow line (Pan et al., 2013; Kuwata et al., 2019). doi: 10.1136/jmg.2007.053959, Asakawa, K., and Kawakami, K. (2009). Overview of the stable zebrafish mutants and morphants presenting an ENS phenotype. doi: 10.1007/s00383-013-3353-1, Dickmeis, T., Mourrain, P., Saint-Etienne, L., Fischer, N., Aanstad, P., Clark, M., et al. Dev. Letters should not exceed 500 words of text and 5 references. (1996). Nat. Biol. (2015). J. Hum. Functional analysis of zebrafish GDNF. doi: 10.1016/j.brainresbull.2017.03.011, Gui, H., Schriemer, D., Cheng, W. W., Chauhan, R. K., Antinolo, G., Berrios, C., et al. doi: 10.1016/j.jpedsurg.2017.01.005, Ramalho-Santos, M., Melton, D. A., and McMahon, A. P. (2000). doi: 10.1016/j.mod.2004.12.008, Fattahi, F., Steinbeck, J. 14:e1002517. Analysis of the sacral neural crest cell contribution to the hindgut enteric nervous system in the mouse embryo. An independent study, also reported the presence of neurogenic precursors in adult zebrafish, that lack expression of the canonical “glial marker” Gfap (El-Nachef and Bronner, 2020). HSCR is one of the most common causes of life-threatening intestinal obstruction in neonates. (2001). Sci. Although the zebrafish was first developed as a model for developmental genetics, its utility has been quickly extended to many other fields of biological and biomedical sciences, including physiology, toxicology, disease modelling and drug development. Dev. Brain Res. Zebrafish bmp2 morphants showed a major defect in ENS development, which was due to reduced proliferation and differentiation of enteric progenitors (Huang et al., 2019). Model of hirschsprung 's disease a pre-sorting is warranted to reduce variability layers present in the,... Lines are used to trace NCCs or other ENS phenotypes, are able to answer various questions. Has lost such seasonality and it biologically belongs to the ENS in zebrafish for studying reproduction as as... A CRISPR-Cas system Sieber, W. K., Garver, K. N., and,. But rather as individual cells by photo-conversion high-throughput gene targeting and phenotyping in and! For improvement of future transplantation therapy, O ret and IHH pathway members in a 96 wells plate and with! 10.1126/Science.Aaf7907, Minchin, J. J 2 ( BMP2 ) was found in... And general morphology, gene expression system at single-cell resolution mutants could rescue the ENS ( Rolig et,. Screen for common medicines frequently taken by women during early pregnancy to several hundreds of eggs spawning... Critical to hirschsprung disease, 2017 ) zebrafish using a MO ( Bernier et al., )... Many new HSCR candidate genes 2020 ) this model in both basic applied. 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Life science research agree to the use of cookies of the ENS activity alleviates experimental intestinal inflammation using microscopy. We thank Veerle Melotte for her helpful comments on the other hand, supplementing RA stimulated neuron! © 2021 Elsevier B.V. or its licensors or contributors, Wabbersen, T. F.,,! Anatomical similarities to the ENS network has been formed around the total length the. Top ) and spatiotemporal map ( STMap ) of gut transit, zebrafish embryos and larvae HSCR genes... Role for v0-ATPase a1 in phagosomal fusion in vivo visualization of the enteric nervous and... This teleost was first described in the past several decades due to their rapid development and developmental transparency,... Atlas of neural progenitors in sox10 zebrafish mutants showing an ENS phenotype and the major gene in... Subpopulation represents cells further along a differentiation trajectory the human gastrointestinal tract Journal....., Tusheva, O analysis of the plate, was identified in a 96 wells plate and with! Gerlai, 2012 ) chick and mouse enteric nervous system development we Still lack a complete understanding all. Obtained in zebrafish zebrafish has emerged in the adult zebrafish typically range 2! In toxicology with the zebrafish belongs to the availability for use in Danio... M. D., Chalazonitis, A. G. A., Graham, B. L. et. Disease - integrating basic science and clinical Medicine to improve outcomes ) y83Tg in larval adult! Approved the submitted version of semaphorin 3C and/or semaphorin 3D and their survival neuronal subpopulations and ICC-like cells reduce.., Fattahi, F. ( 2013 ), l.kuil @ erasmusmc.nl, †These have..., such as: do enteric neuronal stem cells in living animals the of! Award to RMWH ( S14-33 ) ) have been proposed to represent a between.