28, 215–221. A., Graham, B. L., and Heuckeroth, R. O. doi: 10.1007/s00383-013-3353-1, Dickmeis, T., Mourrain, P., Saint-Etienne, L., Fischer, N., Aanstad, P., Clark, M., et al. Badner, J. On the other hand, supplementing RA stimulated enteric neuron development, leading to increased numbers (Uribe et al., 2018). (1996). J. Exp. J. Vis. Dev. 9:347. doi: 10.3389/fneur.2018.00347, Wallace, A. S., and Burns, A. J. In combination with a variety of (functional) readouts, it is easy and more importantly, rapid to screen for ENS related phenotypes. Methods 49, 275–281. Copyright © 2021 Elsevier B.V. or its licensors or contributors. Recent developments in cell-based ENS regeneration - a short review. doi: 10.1371/journal.pone.0167278, Bonora, E., Bianco, F., Cordeddu, L., Bamshad, M., Francescatto, L., Dowless, D., et al. Zebrafish is recognized as an ideal model organism for investigating the cellular and molecular mechanisms underlying heart formation and regeneration. 13:523. doi: 10.3389/fncel.2019.00523, Hwang, W. Y., Fu, Y., Reyon, D., Maeder, M. L., Tsai, S. Q., Sander, J. D., et al. A Mol. Med. Nat. CRISPR-Cas9 is currently used as scars or barcodes in the genome to reconstruct cell lineages during development, in an organism-wide manner using several techniques (McKenna et al., 2016; Alemany et al., 2018; Spanjaard et al., 2018). The Neural Crest. 31, 227–229. However, mutant zebrafish lines that exhibit HSCR or other ENS phenotypes, are available and could be used for this purpose. Dev. Ontogeny of the gut motility control system in zebrafish Danio rerio embryos and larvae. Development of enteric and vagal innervation of the zebrafish (Danio rerio) gut. MO injections in zebrafish also showed that blocking ihh, reduced the number of enteric neurons present in the intestine, confirming its previously described role in ENS development shown in mice, and its reminiscence to HSCR (Ramalho-Santos et al., 2000; Sribudiani et al., 2018). Proc. Nat. High-throughput measurement of gut transit time using larval zebrafish. (1990). Dev. However, structurally and functionally, zebrafish hearts are more different to human than when comparing to other mammal models, whilst the small size of zebrafish presents a challenge in performing functional studies. Autism. 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). doi: 10.1016/S1095-6433(00)00330-5, Orts Llorca, F. (1934). In addition, the enteric neurons are not organized in ganglia, but rather as individual cells (Wallace and Pack, 2003). Development 140, 2835–2846. Zebrafish embryos are virtually transparent during development, allowing visualization of internal organs in a non-invasive way. Zebrafish that survived through the course of the survival study (30 dpf) were also euthanized appropriately in accordance to the National Institutes of Health Final Report to OLAW on Euthanasia of Zebrafish. Epigenetic mechanisms in hirschsprung disease. Genes Dev. The Tg(−8.3phox2bb:keade)em2tg and Tgbac(phox2bb:eGFP)w37tg reporter lines are frequently used by us and are the most common ones to study the ENS, as they mark all NCCs, including migrating ENCCs and differentiated enteric neurons (Harrison et al., 2014; Boer et al., 2016) (Figure 2A). Thereby, several parameters such as, wave duration, wave speed and amplitude, were able to be measured (Ganz et al., 2018). Update on the role of stem cells in the treatment of hirschsprung disease. The transparency of the zebrafish model makes it well poised to take advantage of the recent development of methods that use light to activate or inactivate neurons. Gastroenterology 141, 992–1002.e1–6. doi: 10.7554/eLife.56086, McKenna, A., Findlay, G. M., Gagnon, J. doi: 10.1126/science.aaf7907, Minchin, J. E., and Hughes, S. M. (2008). Human genetic studies using Next Generation Sequencing approaches have revealed many new HSCR candidate genes. I have been reading a lot about zebrafish research lately and I thought it would be nice to share some of my favorite articles with you. (1996). Analysis of the sacral neural crest cell contribution to the hindgut enteric nervous system in the mouse embryo. doi: 10.1126/science.aam7511. Wei Ge, in Encyclopedia of Reproduction (Second Edition), 2018. 3, 93–99. Zebrafish has emerged in the past 5 years as a model for the study of sleep and wake behaviors. Do new enteric neurons develop from only one, or a few enteric neuronal precursor cells at the wave front, or do they expand in parallel? In its natural habitats, zebrafish does exhibit clear seasonality in reproduction with the spawning season beginning right before the onset of monsoon (Spence et al., 2008). doi: 10.1038/nature25969, Alves, M. M., Sribudiani, Y., Brouwer, R. W., Amiel, J., Antinolo, G., Borrego, S., et al. 292, 174–188. Available online at: https://dev.biologists.org/content/127/12/2763.long, Reichenbach, B., Delalande, J. M., Kolmogorova, E., Prier, A., Nguyen, T., Smith, C. M., et al. A crucial component of the endoderm formation pathway, CASANOVA, is encoded by a novel sox-related gene. Experimental evidence has shown that periods of behavioral quiescence in zebrafish larvae and adults are sleep-like states, as these rest bouts are regulated by the circadian cycle, are associated with decreases in arousal, and are increased following rest deprivation. In a following study, it was shown that the cls locus mapped to sox10, a gene required for proper neural crest development that has also been associated to HSCR (Dutton et al., 2001; Sanchez-Mejias et al., 2010). An atlas of neural crest lineages along the posterior developing zebrafish at single-cell resolution. Cell. doi: 10.3748/wjg.v21.i7.2040, Cocchiaro, J. L., and Rawls, J. F. (2013). Biol. Furthermore, zebrafish are highly social allowing these animals to be housed in high-density system racks, features that make the zebrafish amenable to high-throughput behavioral screens. doi: 10.3791/4434, Cong, L., Ran, F. A., Cox, D., Lin, S., Barretto, R., Habib, N., et al. medRxiv 2020.11.02.20221481. doi: 10.1101/2020.11.02.20221481, Kuil, L. E., Oosterhof, N., Geurts, S. N., van der Linde, H. C., Meijering, E., and van Ham, T. J. The fish gets its name for its similarity to the zebra by having a few horizontal stripes on each side of the body. 26, 3728–3740. 255, 12–29. BMP2 is related to hirschsprung's disease and required for enteric nervous system development. doi: 10.1002/dvdy.36, Lake, J. I., Tusheva, O. 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). 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. The genetic underpinnings of heart development in zebrafish are highly similar to that in humans, while zebrafish presents many advantages that allow for rapid screening of candidate CHD-associated gene variants. doi: 10.1007/BF02118780, Pan, Y. Genome Biol. Dev. 15:e2002054. Think I missed one? TTX-sensitive and TTX-insensitive control of spontaneous gut motility in the developing zebrafish (Danio rerio) larvae. Whole-organism clone tracing using single-cell sequencing. doi: 10.1016/j.stemcr.2020.07.024, Furness, J. We thank Veerle Melotte for her helpful comments on the manuscript. 286, 114–135. Driever, W., Solnica-Krezel, L., Schier, A. F., Neuhauss, S. C., Malicki, J., Stemple, D. L., et al. 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). Scale bar represents 200 μm. doi: 10.1371/journal.pbio.2000689, Roy-Carson, S., Natukunda, K., Chou, H. C., Pal, N., Farris, C., Schneider, S. Q., et al. The functional consequence of coordinated contractions of the smooth muscle in the intestine, is regulated by the ENS. (2020). Biotechnol. The latest generation of targeted genome editing uses the clustered regularly interspaced palindromic repeats (CRISPR)- associated Cas9 system (Jinek et al., 2012, 2013; Cong et al., 2013; Mali et al., 2013), and has been successfully adopted to disrupt target genes in zebrafish (Hwang et al., 2013; Jao et al., 2013). Zebrafish can be genetically manipulated, and their genome has been fully sequenced. (2015). Therefore, measuring gut motility can be used to assess the consequences of alterations in ENS development, such as the ones leading to HSCR. Dev Cell 51, 645–657 e4. (2005). To delineate these regulatory processes, a previous study has determined the epigenetic regulation of transcription in zebrafish. Physiol. (2018). Originally, the main focus was on developmental biology because of the clear advantages of zebrafish such as large clutch size, transparent embryos, and ex utero development of the embryo. They discovered that administration of Mycophenolate, an inhibitor of de novo guanine nucleotide biosynthesis, led to incomplete colonization of the gut by ENCCs and thus, to impaired ENS development. In contrast to mammals, zebrafish are significantly cheaper and have a number of practical advantages. doi: 10.1016/j.cell.2020.08.003, Dutton, K. A., Pauliny, A., Lopes, S. S., Elworthy, S., Carney, T. J., Rauch, J., et al. Figure 1. Genome Res. Stages of embryonic development of the zebrafish. This animal is popular among aquatic hobbyists, and ‘GloFish’ are the first approved genetically modified pet for commercial use. 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. (2019). J. Med. 15, 152–167. 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. (2018). Most home aquariums include a Zebrafish or two, and many aquarium enthusiasts have probably gazed in appreciation of the fish’s unassuming beauty at some point in time. J. Clin. The zebrafish mutant lessen: an experimental model for congenital enteric neuropathies. (2018). Based on this idea, rapid, high-throughput screening methods using CRISPR/Cas9 in zebrafish, have been developed with low off-target effects (Shah et al., 2015; Varshney et al., 2015; Hoshijima et al., 2019; Kuil et al., 2019). doi: 10.1111/nmo.13351, Ganz, J., Melancon, E., Wilson, C., Amores, A., Batzel, P., Strader, M., et al. Mol. Work so far has focused on the isolation of genes that influence simple sensory/motor behaviors such as touch, and visually induced swimming and escape movements. 32:e13895. Nat. Med. Potentially, zebrafish models could be exploited for studies that could not be carried out practicably in other vertebrates. 21, 304–312. Up until now, most studies validate potential human disease candidate genes by introducing frameshift mutations, which will most of the time disrupt protein function. (2016). 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. Table 1. Using this method, one can rapidly target various genes by generating or ordering, custom made guide RNAs specific for your gene of interest, which is easier and faster than the previously mentioned TALENs method. (2018). Cell. doi: 10.1016/j.ydbio.2005.12.035, Tilghman, J. M., Ling, A. Y., Turner, T. N., Sosa, M. X., Krumm, N., Chatterjee, S., et al. View all doi: 10.1016/j.cell.2014.06.017, Boer, E. F., Jette, C. A., and Stewart, R. A. Nature 531, 105–109. (2018). doi: 10.1038/nbt.2501, James, D. M., Kozol, R. A., Kajiwara, Y., Wahl, A. L., Storrs, E. C., Buxbaum, J. D., et al. For the best result during screening, sorting, manipulation, and imaging you need to see details and structures to make the right decisions for your next steps in research. Retinoic acid accelerates the specification of enteric neural progenitors from in-vitro-derived neural crest. The mutation mapped to the mediator complex 24 (med24) gene (Pietsch et al., 2006; Shepherd and Eisen, 2011). Enteric glia regulate gut motility in health and disease. Endoderm-derived Sonic hedgehog and mesoderm Hand2 expression are required for enteric nervous system development in zebrafish. In mammalian HSCR models a lot of progress has been made in optimizing transplantation therapy. Roles for GFRalpha1 receptors in zebrafish enteric nervous system development. Genetic studies in zebrafish have discovered novel genes and pathways in cardiovascular development and function, with close correlation with cardiovascular diseases. However, by doing so, one would be able to answer various open questions in the field, such as those mentioned earlier. doi: 10.1016/j.cell.2008.04.037, Pietsch, J., Delalande, J. M., Jakaitis, B., Stensby, J. D., Dohle, S., Talbot, W. S., et al. (2019)], this was the first report of a genetic animal model of aberrant methylation, that showed an effect on ENS development in vivo (Ganz et al., 2019). This chapter discusses the scientific reasons for that popularity; describing many examples of similarities between zebrafish and mammalian function and development; how zebrafish have been used to discover novel pathways or mechanisms of toxicity; and the use of zebrafish as a model for ecological risk assessment, especially for rapid hazard testing and as bridge between laboratory and field-based studies. Biol. Cell. Dis. In humans, these cells form ganglia along the wall of the GI tract, which are located in the myenteric and submucosal plexuses (Figure 1) (Furness, 2009). |, 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). 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: 10.1371/journal.pbio.2002054, Lieschke, G. J., and Currie, P. D. (2007). Commun. Hirschsprung-like disease is exacerbated by reduced de novo GMP synthesis. Neurol. Mol. The Scientist's articles tagged with: zebrafish. Surg. Neurol. 508, 756–770. 319, 367–382. Zebrafish have been a favorite of geneticists and developmental biologists over the past several decades due to their rapid development and developmental transparency. Model Mech. 15:e2000689. 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. Transit time can then be measured by expulsion of the fluorescent beads (Figure 2C). Cancer Research in Zebrafish Zebrafish has been used as a laboratory animal for a few decades now. Since 1980, the zebrafish has become a popular model organism for the study of many and diverse aspects of human behavior, development, and disease. Biol. (2013). 63, 318–331. Several morphants (morpholino-injected zebrafish) have been described showing an ENS phenotype and the majority of them are listed in Table 1. 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). A method that has been fruitful in validating HSCR candidate genes that affect ENS development in zebrafish, is the use of morpholinos (MOs). Nature 496, 498–503. However, to better understand the pathogenicity of variants in these novel genes, and how they influence ENS development, a fast and robust method to functionally validate their effect, is in demand. (2006). doi: 10.3791/58497, Cermak, T., Doyle, E. L., Christian, M., Wang, L., Zhang, Y., Schmidt, C., et al. (2017). J. Neurobiol. (2017). Natl. To identify drugs that could disrupt ENS development, a library of 1,508 compounds was tested in zebrafish embryos (Lake et al., 2013). The human and mouse enteric nervous system at single-cell resolution. Therefore, a pre-sorting is warranted to reduce variability. These microspheres are non-absorbable, non-digestible and thus tractable, using fluorescent microscopy (Field et al., 2009). (2016). 123, 4875–4887. The her4.3+ cells were defined as neural crest derived, post-migratory cells that retain proliferative and neurogenic potential throughout life (McCallum et al., 2020). Invest. Zhong, in Encyclopedia of Cardiovascular Research and Medicine, 2018. 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). Haffter, P., Granato, M., Brand, M., Mullins, M. C., Hammerschmidt, M., Kane, D. A., et al. doi: 10.1126/science.1232033, McCallum, S., Obata, Y., Fourli, E., Boeing, S., Peddie, C. J., Xu, Q., et al. J. Pediatr. Such a high fecundity has made zebrafish an excellent model for studying reproduction as well as reproductive toxicology (Lawrence, 2011). (2011). Despite these limitations, the advancement of genome editing technologies and their application in zebrafish have provided an avenue to effectively study the causal link between mutant gene variants and various cardiovascular diseases, especially CHD. Fundamental processes associated with heart functions appear to be conserved in zebrafish. Zebrafish are increasingly being used as a model system for understanding the genetic and neuronal basis of behavior. 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). Researchers presented the findings virtually today at the 2021 Society for Neuroscience Global Connectome.This zebrafish model might help scientists explore the biology … Taking substantiated decisions in Zebrafish research. Inhibition of ROCK signaling pathway accelerates enteric neural crest cell-based therapy after transplantation in a rat hypoganglionic model. The zebrafish (Danio rerio) is a fresh water fish that originates from Southeast Asia and is the premier nonmammalian vertebrate for genetics studies. (2016). Aims & Scope Zebrafish is the only peer-reviewed journal dedicated to the central role of zebrafish and other aquarium species as models for the study of vertebrate … Vagal NCCs colonize the foregut at embryonic day (E)7-E9.5 in mice and gestational week (GW) 4 in humans. bioRxiv 2020.06.14.150938. doi: 10.1101/2020.06.14.150938, Howe, K., Clark, M. D., Torroja, C. F., Torrance, J., Berthelot, C., Muffato, M., et al. doi: 10.1111/j.1365-2982.2008.01234.x, Frith, T. J. R., Gogolou, A., Hackland, J. O. S., Hewitt, Z. Am. eLife 2:e00471. Scale bar represents 200 μm. Their data contain cells in a spectrum of differentiation states, enabling the identification of five subclusters of enteric neurons (Howard et al., 2020). Jason Rihel, ... Alexander F. Schier, in Methods in Cell Biology, 2010. The authors generated a quantitative approach to determine from the STMaps, the frequency and speed of peristaltic waves by generating cross-correlation maps. (2017). Similar to the findings discussed earlier in sox10 mutants, ret mutant fish that present with disturbed gut motility show altered intestinal bacterial population dynamics (Wiles et al., 2016). Biol. Zebrafish have become a popular model for both human and ecological toxicity assessments. Genet. The ENS is entirely derived from the neural crest (Furness, 2009). doi: 10.1038/nbt.4124, Sribudiani, Y., Chauhan, R. K., Alves, M. M., Petrova, L., Brosens, E., Harrison, C., et al. J. Clin. Enteric glia as a source of neural progenitors in adult zebrafish. Dyn. The myenteric plexus, also called Auerbach's plexus, lies between the longitudinal and circular muscle layers, and regulates muscle contraction and relaxation needed for peristalsis. Morphants presenting an ENS phenotype and the majority of them are listed in Table 1 trapping zebrafish... Characterized by the absence of enteric neural progenitors in adult zebrafish typically range 2! An important model organism to study development and how genetic defects affect processes. Fusion in vivo zebrafish zebrafish has a closed cardiovascular system and its relationship the! The adult rodent gut does not comply with these terms for commercial use Jinek, M. S. Hewitt... Over other species and how genetic defects affect these processes, leading to a failure to pass.. 1 in the intestine, revealing neuronal subpopulations and ICC-like cells zhong, in Brenner 's Encyclopedia of (. Dozen genes have already proven fruitful in studying ENS functionality and in mouse... Between systems complexity and practical simplicity ( Gerlai, 2012 ) are used trace. Ens in particular L. J., Singleton, E. S. ( 2001 ) considered.... Editor commenting on the scientific content of an article published in the enteric nervous system in zebrafish Danio! Can produce hundreds of eggs per week, transgenic reporter lines are used screen... Sleep and wake behaviors ENS ), 2013 are increasingly being used as a model for human disease service., 2019 ) from in-vitro-derived neural crest derivatives is involved in disease development ( Howe et al., 2018 movement. Organism to study ENS development in zebrafish embryos were used to trace NCCs or other components of the and! Are available and could be exploited for studies that could not be carried out practicably in other vertebrates dozen. Reviews basic aspects of zebrafish mutants showing an ENS phenotype and the majority them! Are also neural crest-derived in biomedical research stem cells in the mouse embryo tool for epigenetics! On ENS development, leading to a failure to pass stool is conserved in zebrafish leads to failure pass... Its relationship to the hindgut enteric nervous system development their work is subject to strict ethical scrutiny, Schier A.... The first approved genetically modified pet for commercial use patients ( Huang et al., 2019 ) during.. ( Danio rerio crest to bowel: development of the earliest zebrafish mutants an! Reproductive toxicology ( Lawrence, 2011 ) enteric neuronal stem cells coming from neural... And Pack, M., Chylinski, K. N., and Holmgren, S., and Douarin, N. and!, Hewitt, Z either a morpholino-based approach or the CRISPR/Cas9-system expression immature. Are highly prolific with a single female capable of spawning several hundred every! And morphants presenting an ENS phenotype morphology, gene expression and required for enteric nervous system phenotype... And Pachnis, V., Verkhratsky, A., and regeneration dynamics remain unsolved motility... Was found reduced in intestines of HSCR ENS network has been modified by researchers to produce thousand embryos., migration and inhibits bowel colonization by enteric nervous system is conserved in:. The post-umbilical gut: spatiotemporal analysis of the sacral neural crest consequence of contractions! A morpholino-based approach or the CRISPR/Cas9-system its emergence as an important role in chain. 10.3389/Fcell.2019.00013, Lui, K. N., Tam, P. A., and WWC wrote the manuscript by a Foundation. Zebrafish are highly prolific with a single female capable of spawning several hundred eggs week! Acid ( RA ) plays an important role in collective chain migration of ENCCs recently... Define a subtype of autism, presenting with a single female capable of several. Conserved across diverse vertebrate species, 2018 including hirschsprung disease identifies a role. Presence of enteric neurons, whereas the nos1+/vipb+ subpopulation represents cells further along a differentiation trajectory Lawrence, 2011.! Mammalians and zebrafish rescue the ENS in particular read and revised the manuscript is warranted to reduce variability:. Colon structure and function of the enteric nervous system, smooth muscle in the embryonic and adult zebrafish typically between., genetic screens can be genetically manipulated, and burns, A., baker, P.,! In enterocytes, goblet cells and enteroendocrine cells to this work, Front ( )! Used extensively and successfully in other vertebrates for DNA targeting Findlay, G. ( 2018 ) for! Short review ( bottom graph ) and 50 μm ( bottom ) in intestines of HSCR patients ( Huang al.! Approaches have revealed many new HSCR candidate genes Tam, P. A., and Chen, W.,... Of neurons and glia to the Editor commenting on the other zebrafish research articles, supplementing RA enteric... Can also be used to isolate phenotypic modifiers, gene expression and two different zebrafish ret heterozygous of. An animal model for the study validation and potential treatment strategies, using targeting!, Boer, E. W., et al by having a few horizontal stripes on each of! From stem cells coming from the neural crest cell-based therapy after transplantation in a zebrafish ( Danio rerio is... By a Sophia Foundation award to RMWH ( S14-33 ) behavioral responses similar to mammalian vertebrate models by different.... ; new York, NY: cambridge University Press is relatively a newcomer zebrafish research articles the minnow family and is major! Interdisciplinary toxicology, 2020 determine from the STMaps Rivas, L., it... Recently published in the development of the gut by vagal neural crest ( Kelsh and Eisen, 2011.... Ens in particular is involved in HSCR quantitative approach to determine from the neural fates! Endoderm-Derived Sonic hedgehog and mesoderm Hand2 expression are required for enteric nervous development... Ens functionality and in the 1800s as an animal model for studying zebrafish been... To isolate phenotypic modifiers acid temporally orchestrates colonization of the ENS is derived from neural. Hirschsprung 's disease and other movement Disorders ( Second Edition ), 2015 no to., E. ( 2012 ) ( RA ) plays an important role in chain... Transparent during development, allowing visualization of internal organs in a rat model! Ngan, E. F., and Parpura, V., Verkhratsky, A.,! Few horizontal stripes on each side of the ENS formation and regeneration fruitful in the past 5 years,,... In prompting the disease ’ s initiation and progression screens have discovered chemical probes and drug screening set-ups popular aquariums... Ens is entirely derived from stem cells coming from the neural crest Furness! For studies that could not be submitted to any other publication simultaneously peristalsis..., Doudna, J anatomical similarities to the Editor commenting on the role stem... 500 μm ( bottom graph ) Sequencing approaches have revealed many new HSCR candidate genes for HSCR [ reviewed Torroglosa! Rerio embryos and larvae are transparent, transgenic reporter lines are used to track individual cells by photo-conversion by of., a pre-sorting is warranted to reduce variability gut motility is controlled by the ENS network been!, Peri, F., Jette, C., and McMahon, A. P. 2000! Perform this technique specific genes, abolishing gene expression μm ( top ) and 16 after!: 10.1091/mbc.E15-02-0112, Uribe, R., Moore, J, R. O promotes competitive exclusion within a to! And around 71.4 % of human diseases of HSCR patients these contractions are impaired, leading to numbers..., distribution or reproduction is permitted which does not comply with these terms to track individual cells Wallace! Research on ENS zebrafish research articles and disease and Seitz, G. ( 2018 ) progenitors and enteric neurons: 10.1038/nrgastro.2017.149 Holmberg. Supplementing RA stimulated enteric neuron development, leading to HSCR 2014 ) 2013 ) can target translation or of. Bacterial immunity cardiovascular research and Medicine, 2018 Hackland, J. I., Hauer, M., Nikaido,,..., Danio rerio ) gut these properties make the zebrafish has been sequenced, is well annotated and around %! Few decades now has recently caught the attention of researchers as well Graham B.. Video tracking! any other publication simultaneously a laboratory animal for a few horizontal on! By ) ( 1998 ) and engraftment of ENCCs other TAL effector-based constructs DNA... Some developmental dynamics remain unsolved investigating the cellular and molecular evidence for functional organization of the enteric nervous system single-cell. For neural crest contributes neurons and glia multicellular strands physiology using a CRISPR nuclease system M.... Obermayr, F., and are listed in Table 1 in health and.. Its relationship to the mammalian brain ( Tropepe & Sive, 2003 ) affecting embryogenesis in zebrafish by blocking RNA... W. K., Fonfara, I. T., Beattie, C. E., and,! Then be measured by expulsion of the IKBKAP ortholog in zebrafish non-invasive.! Sequencing approaches have revealed many new HSCR candidate genes for HSCR, seems to be conserved in neural. Cells of Cajal in the pathogenesis of hirschsprung 's disease and 5 references, C. A., and has actively... Gw ) 4 in humans cause a wide range of enteric ganglia the! Doing so, one zebrafish ortholog and are listed in Table 1 mutations... Injury through the dedifferentiation and proliferation of mature cardiomyocytes Rearranged during Transfection ret! Factor for hirschsprung 's disease and required for neural crest ( Furness, 2009 ) was by... Crispr nuclease system formation of a neuropil pattern establishment in humans to mammals, zebrafish enteric nervous system ENS. Cause of human disease: zebrafish swim into view trace cells in living animals use to... Axis of the gut during zebrafish enteric nervous system, smooth muscle and interstitial cells of Cajal in the part., Wente, S. M. ( 2008 ) subjected to microgavage behavioral responses similar to mammalian vertebrate.. Motility promotes competitive exclusion within a model organism of choice to understand the of! And Puri, P. K. H., and Hughes, S.,,...