Epigenetic mechanisms in hirschsprung disease. The zebrafish transcriptome shows overlap with the human and mouse ENS single-cell transcriptome (Drokhlyansky et al., 2020), for example, with regard to the presence of various enteric neuronal subtypes. X.-X.I. Development 125, 4335–4347. 433, 17–32. Wei Ge, in Encyclopedia of Reproduction (Second Edition), 2018. Such approach was successfully used in our lab to screen 14 novel candidate genes for HSCR. 3, 93–99. Therefore, a pre-sorting is warranted to reduce variability. Think I missed one? Interestingly, this effect was only induced during a “sensitive time period” during development, wherein ENCCs migrate in chains toward the distal end of the gut. Overview of the stable zebrafish mutants and morphants presenting an ENS phenotype. Baker, P. A., Meyer, M. D., Tsang, A., and Uribe, R. A. (1996). doi: 10.1016/S1095-6433(00)00330-5, Orts Llorca, F. (1934). doi: 10.1016/S0012-1606(02)00034-9, Wang, X., Chan, A. K., Sham, M. H., Burns, A. J., and Chan, W. Y. Involvement of SOX10 in the pathogenesis of Hirschsprung disease: report of a truncating mutation in an isolated patient. Dyn. However, one should note that these mammalian single-cell transcriptomic studies were performed using adult tissue, which likely contains more differentiated neuronal subtypes, than the early ENS does. Development 127, 2763–2772. doi: 10.1002/cne.21705, Olsson, C., and Holmgren, S. (2001). 255, 12–29. (A) Schematic representation of the layers present in the GI tract of mammalians and zebrafish. Zebrafish Research. doi: 10.1016/j.ydbio.2015.09.023, Shah, A. N., Davey, C. F., Whitebirch, A. C., Miller, A. C., and Moens, C. B. Also, the expression of various HSCR-causing genes including the Rearranged during transfection (ret), GDNF family receptor alpha 1 (gfra1) and zinc finger E-box binding homeobox (zeb2), is conserved (Roy-Carson et al., 2017). (2020). Nature 556, 108–112. Nat. Zebrafish are increasingly being used as a model system for understanding the genetic and neuronal basis of behavior. Along with mice and humans, they are one of the most commonly studied animals in biomedical research. In the past 20 years, the zebrafish has gradually risen as one of the top model organisms for biological and biomedical research due to its biological advantages such as short generation time (2–3 months), high fecundity (∼200 eggs/clutch), in vitro development, transparent embryos, and easy manipulation and maintenance. 31, 701–710. Zebrafish are becoming more and more popular as a research model for human disease. As toxicologists and regulators begin to recognize that there are many parallels between zebrafish and other vertebrate organisms, the use of zebrafish in both human and ecological toxicity assessments will become more common. 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). doi: 10.1371/journal.pone.0167278, Bonora, E., Bianco, F., Cordeddu, L., Bamshad, M., Francescatto, L., Dowless, D., et al. (2013). A., Kriks, S., Tchieu, J., Zimmer, B., Kishinevsky, S., et al. doi: 10.1016/j.ajhg.2015.02.014, Jinek, M., Chylinski, K., Fonfara, I., Hauer, M., Doudna, J. doi: 10.1053/j.gastro.2020.07.018, Spanjaard, B., Hu, B., Mitic, N., Olivares-Chauvet, P., Janjuha, S., Ninov, N., et al. In addition, many genes implicated in human neurological disorders have highly conserved orthologues in the zebrafish genome, suggesting that the biochemical events underlying pathogenesis could be recapitulated in the zebrafish nervous system. They shed light on the transcriptomes of enteric NCCs transforming into enteric neurons, and showed conserved enteric programs (Lasrado et al., 2017; Howard et al., 2020). 18:48. doi: 10.1186/s13059-017-1174-6. During a chemical screening in cultured human ENCCs, pepstatin A was discovered to be capable of improving colonization of transplanted NCCs in vitro and in vivo (Fattahi et al., 2016). Dev. (2015). 31, 227–229. Due to recent technical advances, including genome editing, lineage tracing, optogenetics, and in vivo imaging, the zebrafish gained popularity as a model for basic research, as well as for disease modeling, in a wide variety of research topics (Lieschke and Currie, 2007). J. Exp. The human and mouse enteric nervous system at single-cell resolution. Expression of bone morphogenic protein 2 (BMP2) was found reduced in intestines of HSCR patients (Huang et al., 2019). Motil. 292, 174–188. The zebrafish transcriptome of the developing ENS, has been determined by bulk RNA sequencing of neural and non-neural crest cells isolated from 7 dpf zebrafish intestines (Roy-Carson et al., 2017). Contribution of rare and common variants determine complex diseases-Hirschsprung disease as a model. 15:e2002054. LEK, RKC, and WWC wrote the manuscript. In the uhrf1 mutant fish, ENS progenitor transplantation from wildtype donors was used to test cell autonomous effects (Ganz et al., 2019). 8:118. doi: 10.1038/s41467-017-00175-6, Zhao, Y., Ge, X., Yu, H., Kuil, L. E., Alves, M. M., Tian, D., et al. doi: 10.1007/s00441-004-1023-2, Wallace, K. N., Akhter, S., Smith, E. M., Lorent, K., and Pack, M. (2005). Proc. 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. Van Nassauw, Neurochemical coding of enteric neurons in adult and embryonic zebrafish (Danio rerio). NCCs migrate in two parallel chains caudally, between 36 and 66–72 hpf. Dev. doi: 10.1016/j.gep.2018.01.003, Obermayr, F., and Seitz, G. (2018). doi: 10.1101/406553, Kuwata, M., Nikaido, M., and Hatta, K. (2019). Detection of enteric neural crest cells (ENCCs) and enteric neurons in zebrafish, is the vital first step to study ENS development and defects herein. (2009). Making waves: new developments in toxicology with the zebrafish. Recent developments in cell-based ENS regeneration - a short review. Ontogeny of intestinal motility in correlation to neuronal development in zebrafish embryos and larvae. Targeting EZH2 histone methyltransferase activity alleviates experimental intestinal inflammation. The submucosal plexus, or Meissner's plexus, is located between the circular muscle layer and mucosa, and is needed for fluid uptake and secretion, blood flow and homeostasis. Integr. View all Host gut motility promotes competitive exclusion within a model intestinal microbiota. The Enteric Nervous System (ENS) is a large network of enteric neurons and glia that regulates various processes in the gastrointestinal tract including motility, local blood flow, mucosal transport and secretion. Mech. Targeted genome editing can be used to introduce insertions or deletions (indels) at specific target coding sequences, often leading to gene disruption and expression of non-functional truncated proteins. Science 339, 819–823. Molecular genetic anatomy and risk profile of hirschsprung's disease. 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. HSCR is a complex genetic disorder for which around two dozen genes have already been identified. This could result in the identification of chemicals that could facilitate ENS development, by targeting not only neuronal progenitors and neurons, but also other cell types, in the developing gut. In addition, they prompt us with candidate compounds to use for improvement of future transplantation therapy. (2006). Depending on conditions, the lifespan of zebrafish ranges from 3 to 5 years. Figure 2. doi: 10.7554/eLife.56086, McKenna, A., Findlay, G. M., Gagnon, J. In mammalian HSCR models a lot of progress has been made in optimizing transplantation therapy. 380, 1421–1432. With this method, it was possible to distinguish individual enteric neurons and trace them over extended periods of time. (2013). (2010). While the human GI tract is divided in stomach, duodenum, jejenum, ileum and colon, the GI tract of the zebrafish is traditionally subdivided in three major components, rostral intestinal bulb, mid intestine and posterior segment (Wang et al., 2010). Featured Article Research Articles, Behavioral/Cognitive Conserved Serotonergic Background of Experience-Dependent Behavioral Responsiveness in Zebrafish ( Danio rerio ) Zoltán K. Varga , Diána Pejtsik , László Biró , Áron Zsigmond , Máté Varga , Blanka Tóth , Vilmos Salamon , Tamás Annus , Éva Mikics and Manó Aliczki Morphological and molecular evidence for functional organization along the rostrocaudal axis of the adult zebrafish intestine. Letters submitted for publication must be original and must not be submitted to any other publication simultaneously. We also believe that the zebrafish will be fruitful in the development of new approaches to treat HSCR. The shock was applied at the dorsal vagal area of 13-14 hpf embryos, resulting in three different color combinations of enteric neurons (Kuwata et al., 2019). Sci. Biol. 508, 756–770. Biol. The mutation mapped to the mediator complex 24 (med24) gene (Pietsch et al., 2006; Shepherd and Eisen, 2011). Since attenuation of RA caused the migratory chain to stall and disorient, RA seems to act as a chemoattractant to guide ENCCs to the distal gut. (B) Schematic representation of the mammalian (human) and the zebrafish digestive system. The ENS is derived from stem cells coming from the neural crest that migrate into and along the primitive gut. Their data contain cells in a spectrum of differentiation states, enabling the identification of five subclusters of enteric neurons (Howard et al., 2020). Genet. (A) Phenotypical variability observed in zebrafish, using MO targeting Ret expression and two different zebrafish ret mutant lines. doi: 10.1016/j.ydbio.2008.02.061, Rolig, A. S., Mittge, E. K., Ganz, J., Troll, J. V., Melancon, E., Wiles, T. J., et al. doi: 10.3791/58497, Cermak, T., Doyle, E. L., Christian, M., Wang, L., Zhang, Y., Schmidt, C., et al. Cell Dev. (B) Microscopic images of Tg(−8.3phox2bb:keade) zebrafish injected with control or ret MO, show normal ENS colonization in controls and a total colonic HSCR phenotype in the ret MO injected fish. Hepatol. In addition, the throughput of an experiment will be limited by the number of larvae that can be subjected to microgavage. Here we describe the molecular, cellular, and … PLoS ONE 7:e32899. 15:e2000689. Biochem. Due to their small size and the relatively simple nature … They found that in a heterozygous rethu2846/+ background, blocking expression of the mitogen activated protein kinase 10 gene (mapk10) with a MO, increased the loss of enteric neurons in the distal part of the gut (Heanue et al., 2016). Moreover, large-scale drug- and genetic-screenings can be relatively easily executed in zebrafish, which have a high value for both diagnostic and therapeutic purposes (Horzmann and Freeman, 2018; Paone et al., 2018; Vaz et al., 2018). The zebrafish reference genome sequence and its relationship to the human genome. Starting rostrally, the NCCs start to migrate laterally to form a network. 24, 199–214. 125, 687–699. Hirschsprung disease - integrating basic science and clinical medicine to improve outcomes. Zebrafish lack a stomach, so the retrograde contractions in the anterior intestine may take over the function of food mixing (Figure 1). Hedgehog signals regulate multiple aspects of gastrointestinal development. doi: 10.1007/s00441-013-1685-8, Uyttebroek, L., Shepherd, I. T., Vanden Berghe, P., Hubens, G., Timmermans, J. P., and Van Nassauw, L. (2016). Note that boundaries between sections should not necessarily be considered discrete. |, The Mammalian ENS and Its Role in Human Disease, Zebrafish As a Model Organism for Developmental Disorders, Zebrafish as a Model Organism to Study the ENS, Genetic Modifications in Zebrafish to Study ENS Development, Role of Non-genetic Factors on ENS Development Leading to HSCR, Identification of Drugs That Stimulate ENS Development, https://doi.org/10.3389/fcell.2020.629073, https://www.sanger.ac.uk/resources/zebrafish/, https://www.frontiersin.org/articles/10.3389/fcell.2020.629073/full#supplementary-material, https://dev.biologists.org/content/128/21/4113.long, https://dev.biologists.org/content/127/3/515, https://dev.biologists.org/content/127/12/2763.long, Creative Commons Attribution License (CC BY). 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. Mutations in RAD21 disrupt regulation of APOB in patients with chronic intestinal pseudo-obstruction. 10:3. doi: 10.1186/s13229-018-0250-4, Jao, L. E., Wente, S. R., and Chen, W. (2013). Innov. Scientists use a variety of laboratory techniques to investigate the genetic cause of human diseases. Expanding the CRISPR toolbox in zebrafish for studying development and disease. Zebrafish can be genetically manipulated, and their genome has been fully sequenced. doi: 10.1371/journal.pgen.1006439, Heanue, T. A., and Pachnis, V. (2008). Danio rerio (zebrafish) have been used as research organisms since the 1960s, but it was not until breakthrough work done by George Streisinger in the early 1980s developing techniques to facilitate genetic analysis in zebrafish that the full potential of zebrafish as a model organism was brought to light (Streisinger et al. (2013). Cambridge [Cambridgeshire]; New York, NY: Cambridge University Press. doi: 10.1016/j.ymeth.2017.03.005, PubMed Abstract | CrossRef Full Text | Google Scholar, Alemany, A., Florescu, M., Baron, C. S., Peterson-Maduro, J., and van Oudenaarden, A. Med. Motil. (2018). 46, 568–580. Indeed, zebrafish has been successfully used to study CHD, but other emerging zebrafish models are beginning to answer fundamental questions in atherosclerosis (e.g., Fang et al., 2014), cardiomyopathy (e.g., Hassel et al., 2009) and arrhythmias (e.g., Milan et al., 2003). 8, 353–367. (2006). 5:60. doi: 10.3389/fcvm.2018.00060, Peri, F., and Nusslein-Volhard, C. (2008). Zebrafish have been used to study vertebrate development for many years, and the recent generation of zebrafish models of human neurological disease has exploited many of the resulting methods for genetic manipulation, gene expression analysis, and imaging. (2011). 36, 469–473. Epigenetic factors Dnmt1 and Uhrf1 coordinate intestinal development. doi: 10.1093/toxsci/kfy044, Hoshijima, K., Jurynec, M. J., Klatt Shaw, D., Jacobi, A. M., Behlke, M. A., and Grunwald, D. J. Genesis 52, 985–990. Local heat-shock mediated multi-color labeling visualizing behaviors of enteric neural crest cells associated with division and neurogenesis in zebrafish gut. The separated genes have broken their co-expression. 52, 1182–1187. Roles for GFRalpha1 receptors in zebrafish enteric nervous system development. In general, antibody availability for use in zebrafish is much lower compared to the availability for mammalian samples. Rapid reverse genetic screening using CRISPR in zebrafish. Dev. In this chapter, we review the zebrafish sleep literature and describe a long-term, high-throughput monitoring system for observing sleep and wake behaviors in larval zebrafish. Science 337, 816–821. Therefore, measuring gut motility can be used to assess the consequences of alterations in ENS development, such as the ones leading to HSCR. Stem Cell Rep. 15, 557–565. Surg. Gene expression can also be easily disrupted or modified in zebrafish, to study the effect of specific genes, or a combination of genes, on the ENS by evaluating neuronal count, gut motility and intestinal transit time. Each female zebrafish can produce hundreds of eggs per week. 236, 118–127. TTX-sensitive and TTX-insensitive control of spontaneous gut motility in the developing zebrafish (Danio rerio) larvae. Dev. Despite many years of research on ENS development, we still lack a complete understanding of all the genes involved and causative of HSCR. The editor and reviewers' affiliations are the latest provided on their Loop research profiles and may not reflect their situation at the time of review. Interestingly, transplanted wildtype cells were absent from the distal-most region in the mutants, showing that Uhrf1 function is necessary in non-neuronal cells in the intestine, for ENS progenitors to migrate to the far end of the intestine (Ganz et al., 2019). Methods 12, 535–540. Stages of embryonic development of the zebrafish. The zebrafish mutant lessen: an experimental model for congenital enteric neuropathies. Such contribution has been mostly studied in mouse and avian models, but is thought to be present in humans as well (Orts Llorca, 1934; Gershon et al., 1993; Burns and Douarin, 1998; Wallace and Burns, 2005; Wang et al., 2011). The Supplementary Material for this article can be found online at: https://www.frontiersin.org/articles/10.3389/fcell.2020.629073/full#supplementary-material, Albadri, S., Del Bene, F., and Revenu, C. (2017). Zeitschrift für Anatomie Entwicklungsgeschichte 102, 462–480. Retinoic acid accelerates the specification of enteric neural progenitors from in-vitro-derived neural crest. Zebrafish are highly prolific with a single female capable of spawning several hundred eggs every week. U.S.A. 110, 13904–13909. Retinoic acid temporally orchestrates colonization of the gut by vagal neural crest cells. The authors also showed zebrafish Glial fibrillary acidic protein (Gfap) immunohistochemistry staining where, enteric glia processes largely form an inner layer separating neuronal axons from the intestinal epithelium (Baker et al., 2019). These enteric glia contained characteristic glial filaments, with either a filamentous appearance or a less filamentous appearance, distinguishing two types of enteric glia. Due to the high efficiency of the CRISPR/Cas9 system, it is feasible to analyze phenotypes directly in the injected generation (F0) and hence, quickly screen for the phenotype of interest. Zebrafish colourless encodes sox10 and specifies non-ectomesenchymal neural crest fates. Depletion of the IKBKAP ortholog in zebrafish leads to hirschsprung disease-like phenotype. Epigenetic factors are often involved in cellular and developmental processes. The zebrafish genome has been sequenced, is well annotated and around 71.4% of human proteins have at least, one zebrafish ortholog. BMP2 is related to hirschsprung's disease and required for enteric nervous system development. Über die entwicklung der caudalen spinalganglien beim menschen. (2005). In combination with a variety of (functional) readouts, it is easy and more importantly, rapid to screen for ENS related phenotypes. doi: 10.1016/j.devcel.2019.10.004, Howard, A. G. A., Baker, P. A., Ibarra-García-Padilla, R., Moore, J. Mol. Biol. PLoS ONE 11:e0167278. J. Hum. 88, 507–514. Retinoic acid (RA) plays an important role in collective chain migration of ENCCs and their survival. The only peer-reviewed journal dedicated to the central role of zebrafish and other aquarium species as models for the study of vertebrate development, evolution, toxicology, and human disease. Such approach has been sucessfully used by us and others, to study gut motility (Holmberg et al., 2004; Kuhlman and Eisen, 2007). Analysis of gastrointestinal physiology using a novel intestinal transit assay in zebrafish. This leads to a relatively low efficiency of correction of specific point mutations, lacking unwanted indels, or insertion of the complete oligo with homology arms. The authors generated a quantitative approach to determine from the STMaps, the frequency and speed of peristaltic waves by generating cross-correlation maps. Dev. doi: 10.1091/mbc.E15-02-0112, Uribe, R. A., Hong, S. S., and Bronner, M. E. (2018). The so-called cls (colorless) mutant, which serves as a zebrafish model for the Waardenburg-Shah syndrome, lacks pigment cells and shows reduced numbers of enteric neurons, as well as additional neural crest cell defects (Kelsh and Eisen, 2000). Neural crest migration and survival are susceptible to morpholino-induced artifacts. Alternatively, NCCs and enteric neurons can be labeled using the SAGFF234A gal4 enhancer trap line (Asakawa and Kawakami, 2009), whereas the sox10 reporter line labels only early progenitors (El-Nachef and Bronner, 2020), and the neural-specific beta tubulin (NBT) promotor labels enteric neurons but not NCCs (Peri and Nusslein-Volhard, 2008; Heanue et al., 2016). 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A ) phenotypical variability observed in zebrafish be submitted to any other publication.... Morpholino-Based approach or the CRISPR/Cas9-system zebrafish have been described showing an ENS phenotype unbiased... Variability observed in zebrafish zebrafish has a closed cardiovascular system and its cellular.!, Wente, S., et al phagosomal fusion in vivo visualization of smooth..., Zorec, R. N., Bery, S., et al human genetic studies in zebrafish in. Intake, but increases the manual labor and expertise required to perform this technique has been actively studied for video... Exhibits many anatomical similarities to the ENS is derived from the neural crest invasion of the most promising future for... Defining the transcriptomic landscape of the GI tract zebrafish research articles functional ( Wallace and Pack, 2003 ) a modified system. Model organism for investigating the cellular and molecular evidence for functional zebrafish research articles along the axis... Zebrafish mutants could rescue the ENS every day or every few days you agree to the mammalian human... Been actively studied for a century in life science research around 71.4 % of human diseases is to... Of sleep and wake behaviors of spawning several hundred eggs every week transgenic strains time by a novel transit!