{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T19:23:22Z","timestamp":1790364202727,"version":"4.1.0"},"reference-count":104,"publisher":"Portland Press Ltd.","issue":"1","license":[{"start":{"date-parts":[[2024,1,4]],"date-time":"2024-01-04T00:00:00Z","timestamp":1704326400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["portlandpress.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,2,28]]},"abstract":"<jats:p>The enteric nervous system (ENS) is a complex series of interconnected neurons and glia that reside within and along the entire length of the gastrointestinal tract. ENS functions are vital to gut homeostasis and digestion, including local control of peristalsis, water balance, and intestinal cell barrier function. How the ENS develops during embryological development is a topic of great concern, as defects in ENS development can result in various diseases, the most common being Hirschsprung disease, in which variable regions of the infant gut lack ENS, with the distal colon most affected. Deciphering how the ENS forms from its progenitor cells, enteric neural crest cells, is an active area of research across various animal models. The vertebrate animal model, zebrafish, has been increasingly leveraged to understand early ENS formation, and over the past 20 years has contributed to our knowledge of the genetic regulation that underlies enteric development. In this review, I summarize our knowledge regarding the genetic regulation of zebrafish enteric neuronal development, and based on the most current literature, present a gene regulatory network inferred to underlie its construction. I also provide perspectives on areas for future zebrafish ENS research.<\/jats:p>","DOI":"10.1042\/bst20230343","type":"journal-article","created":{"date-parts":[[2024,1,4]],"date-time":"2024-01-04T07:17:58Z","timestamp":1704352678000},"page":"177-190","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["Genetic regulation of enteric nervous system development in zebrafish"],"prefix":"10.1042","volume":"52","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0427-4493","authenticated-orcid":false,"given":"Rosa A.","family":"Uribe","sequence":"first","affiliation":[{"name":"1Biosciences Department, Rice University, Houston, TX 77005, U.S.A."},{"name":"2Laboratory of Neural Crest and Enteric Nervous System Development, Rice University, Houston, TX 77005, U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2024,1,4]]},"reference":[{"key":"2024022816503608500_BST-2023-0343CC1","volume-title":"Neuroscience","year":"2001","edition":"2nd edn"},{"key":"2024022816503608500_BST-2023-0343CC2","volume-title":"The Enteric Nervous System","year":"2006"},{"key":"2024022816503608500_BST-2023-0343CC3","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1242\/dev.30.1.31","article-title":"The migration of neural crest cells to the wall of the digestive tract in avian embryo","volume":"30","year":"1973","journal-title":"J. Embryol. Exp. Morphol."},{"key":"2024022816503608500_BST-2023-0343CC4","doi-asserted-by":"publisher","first-page":"236","DOI":"10.1002\/aja.1002010307","article-title":"Mapping the origin of the avian enteric nervous system with a retroviral marker","volume":"201","year":"1994","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC5","doi-asserted-by":"publisher","first-page":"349","DOI":"10.1242\/dev.122.1.349","article-title":"Common origin and developmental dependence on c-ret of subsets of enteric and sympathetic neuroblasts","volume":"122","year":"1996","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC6","doi-asserted-by":"publisher","first-page":"1894","DOI":"10.1038\/s41467-021-22146-8","article-title":"Clonal analysis and dynamic imaging identify multipotency of individual Gallus gallus caudal hindbrain neural crest cells toward cardiac and enteric fates","volume":"12","year":"2021","journal-title":"Nat. Commun."},{"key":"2024022816503608500_BST-2023-0343CC7","doi-asserted-by":"publisher","first-page":"52","DOI":"10.1002\/wdev.8","article-title":"Neural crest specification: tissues, signals, and transcription factors","volume":"1","year":"2012","journal-title":"WIREs Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC8","doi-asserted-by":"publisher","first-page":"S98","DOI":"10.1016\/j.ydbio.2018.07.004","article-title":"Migration and diversification of the vagal neural crest","volume":"444","year":"2018","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC9","doi-asserted-by":"publisher","first-page":"715","DOI":"10.1016\/j.tig.2017.07.015","article-title":"Regulatory logic underlying diversification of the neural crest","volume":"33","year":"2017","journal-title":"Trends Genet."},{"key":"2024022816503608500_BST-2023-0343CC10","doi-asserted-by":"publisher","first-page":"11","DOI":"10.1007\/s00441-005-0047-6","article-title":"Phenotypes of neural-crest-derived cells in vagal and sacral pathways","volume":"323","year":"2006","journal-title":"Cell Tissue Res."},{"key":"2024022816503608500_BST-2023-0343CC11","doi-asserted-by":"publisher","first-page":"455","DOI":"10.1016\/j.ydbio.2004.03.015","article-title":"Dynamics of neural crest-derived cell migration in the embryonic mouse gut","volume":"270","year":"2004","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC12","doi-asserted-by":"publisher","first-page":"e64077","DOI":"10.1371\/journal.pone.0064077","article-title":"Retinoic acid upregulates ret and induces chain migration and population expansion in vagal neural crest cells to colonise the embryonic gut","volume":"8","year":"2013","journal-title":"PLoS One"},{"key":"2024022816503608500_BST-2023-0343CC13","doi-asserted-by":"publisher","first-page":"553","DOI":"10.1016\/j.ydbio.2006.10.017","article-title":"Cell proliferation drives neural crest cell invasion of the intestine","volume":"302","year":"2007","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC14","doi-asserted-by":"publisher","first-page":"1193","DOI":"10.1111\/j.1582-4934.2009.00813.x","article-title":"Development of enteric neuron diversity","volume":"13","year":"2009","journal-title":"J. Cell. Mol. Med."},{"key":"2024022816503608500_BST-2023-0343CC15","doi-asserted-by":"publisher","first-page":"198","DOI":"10.1016\/j.ydbio.2016.07.008","article-title":"Genetics of enteric neuropathies","volume":"417","year":"2016","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC16","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1136\/jmg.2007.053959","article-title":"Hirschsprung disease, associated syndromes and genetics: a review","volume":"45","year":"2008","journal-title":"J. Med. Genet."},{"key":"2024022816503608500_BST-2023-0343CC17","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1111\/cge.12032","article-title":"The developmental genetics of Hirschsprung's disease","volume":"83","year":"2013","journal-title":"Clin. Genet."},{"key":"2024022816503608500_BST-2023-0343CC18","doi-asserted-by":"publisher","first-page":"963","DOI":"10.1172\/JCI10828","article-title":"Pax3 is required for enteric ganglia formation and functions with Sox10 to modulate expression of c-ret","volume":"106","year":"2000","journal-title":"J. Clin. Invest."},{"key":"2024022816503608500_BST-2023-0343CC19","doi-asserted-by":"publisher","first-page":"366","DOI":"10.1038\/20700","article-title":"The homeobox gene Phox2b is essential for the development of autonomic neural crest derivatives","volume":"399","year":"1999","journal-title":"Nature"},{"key":"2024022816503608500_BST-2023-0343CC20","doi-asserted-by":"publisher","first-page":"60","DOI":"10.1038\/ng0198-60","article-title":"SOX10 mutation disrupts neural crest development in Dom Hirschsprung mouse model","volume":"18","year":"1998","journal-title":"Nat. Genet."},{"key":"2024022816503608500_BST-2023-0343CC21","doi-asserted-by":"publisher","first-page":"5151","DOI":"10.1242\/dev.129.22.5151","article-title":"Requirement of signalling by receptor tyrosine kinase RET for the directed migration of enteric nervous system progenitor cells during mammalian embryogenesis","volume":"129","year":"2002","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC22","doi-asserted-by":"publisher","first-page":"5503","DOI":"10.1242\/dev.01421","article-title":"Mice expressing a dominant-negative Ret mutation phenocopy human Hirschsprung disease and delineate a direct role of Ret in spermatogenesis","volume":"131","year":"2004","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC23","doi-asserted-by":"publisher","first-page":"241","DOI":"10.1242\/dev.00912","article-title":"Roles for GFR\u03b11 receptors in zebrafish enteric nervous system development","volume":"131","year":"2004","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC24","doi-asserted-by":"publisher","first-page":"268","DOI":"10.1002\/dvdy.24597","article-title":"Gut feelings: studying enteric nervous system development, function, and disease in the zebrafish model system","volume":"247","year":"2018","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC25","doi-asserted-by":"publisher","first-page":"629073","DOI":"10.3389\/fcell.2020.629073","article-title":"Zebrafish: a model organism for studying enteric nervous system development and disease","volume":"8","year":"2021","journal-title":"Front. Cell Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC26","doi-asserted-by":"publisher","first-page":"4619","DOI":"10.1242\/dev.02668","article-title":"A direct role for Sox10 in specification of neural crest-derived sensory neurons","volume":"133","year":"2006","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC27","doi-asserted-by":"publisher","first-page":"659","DOI":"10.1016\/j.mod.2004.12.008","article-title":"Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent","volume":"122","year":"2005","journal-title":"Mech. Dev."},{"key":"2024022816503608500_BST-2023-0343CC28","doi-asserted-by":"publisher","first-page":"420","DOI":"10.1006\/dbio.2000.0145","article-title":"Functional analysis of zebrafish GDNF","volume":"231","year":"2001","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC29","doi-asserted-by":"publisher","first-page":"e1006439","DOI":"10.1371\/journal.pgen.1006439","article-title":"A novel zebrafish ret heterozygous model of Hirschsprung disease identifies a functional role for mapk10 as a modifier of enteric nervous system phenotype severity","volume":"12","year":"2016","journal-title":"PLoS Genet."},{"key":"2024022816503608500_BST-2023-0343CC30","doi-asserted-by":"publisher","first-page":"dev200668","DOI":"10.1242\/dev.200668","article-title":"In toto imaging of early enteric nervous system development reveals that gut colonization is tied to proliferation downstream of Ret","volume":"149","year":"2022","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC31","doi-asserted-by":"publisher","first-page":"484","DOI":"10.1002\/dvg.20429","article-title":"Differentiation of the zebrafish enteric nervous system and intestinal smooth muscle","volume":"46","year":"2008","journal-title":"Genesis"},{"key":"2024022816503608500_BST-2023-0343CC32","doi-asserted-by":"publisher","first-page":"3728","DOI":"10.1091\/mbc.E15-02-0112","article-title":"Meis3 is required for neural crest invasion of the gut during zebrafish enteric nervous system development","volume":"26","year":"2015","journal-title":"Mol. Biol. Cell"},{"key":"2024022816503608500_BST-2023-0343CC33","doi-asserted-by":"publisher","first-page":"687","DOI":"10.1016\/j.mod.2008.04.006","article-title":"Ret isoform function and marker gene expression in the enteric nervous system is conserved across diverse vertebrate species","volume":"125","year":"2008","journal-title":"Mech. Dev."},{"key":"2024022816503608500_BST-2023-0343CC34","doi-asserted-by":"publisher","first-page":"174","DOI":"10.1016\/j.ydbio.2005.12.035","article-title":"Zebrafish foxd3 is selectively required for neural crest specification, migration and survival","volume":"292","year":"2006","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC35","doi-asserted-by":"publisher","first-page":"1081","DOI":"10.1002\/dvdy.24438","article-title":"Molecular fingerprinting delineates progenitor populations in the developing zebrafish enteric nervous system","volume":"245","year":"2016","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC36","doi-asserted-by":"publisher","first-page":"e60005","DOI":"10.7554\/eLife.60005","article-title":"An atlas of neural crest lineages along the posterior developing zebrafish at single-cell resolution","volume":"10","year":"2021","journal-title":"eLife"},{"key":"2024022816503608500_BST-2023-0343CC37","doi-asserted-by":"publisher","first-page":"803370","DOI":"10.3389\/fcell.2021.803370","article-title":"Elevated Hoxb5b expands vagal neural crest pool and blocks enteric neuronal development in zebrafish","volume":"9","year":"2022","journal-title":"Front. Cell Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC38","doi-asserted-by":"publisher","first-page":"985","DOI":"10.1002\/dvg.22826","article-title":"In vivo visualization of the development of the enteric nervous system using a Tg(\u22128.3bphox2b:Kaede) transgenic zebrafish","volume":"52","year":"2014","journal-title":"Genesis"},{"key":"2024022816503608500_BST-2023-0343CC39","doi-asserted-by":"publisher","first-page":"52","DOI":"10.1016\/j.ydbio.2008.02.061","article-title":"Endoderm-derived Sonic hedgehog and mesoderm Hand2 expression are required for enteric nervous system development in zebrafish","volume":"318","year":"2008","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC40","doi-asserted-by":"publisher","first-page":"e56086","DOI":"10.7554\/eLife.56086","article-title":"Enteric glia as a source of neural progenitors in adult zebrafish","volume":"9","year":"2020","journal-title":"eLife"},{"key":"2024022816503608500_BST-2023-0343CC41","doi-asserted-by":"publisher","first-page":"107070","DOI":"10.1016\/j.isci.2023.107070","article-title":"Unbiased characterization of the larval zebrafish enteric nervous system at a single cell transcriptomic level","volume":"26","year":"2023","journal-title":"iScience"},{"key":"2024022816503608500_BST-2023-0343CC42","doi-asserted-by":"publisher","first-page":"290","DOI":"10.1186\/s12864-017-3653-2","article-title":"Defining the transcriptomic landscape of the developing enteric nervous system and its cellular environment","volume":"18","year":"2017","journal-title":"BMC Genomics"},{"key":"2024022816503608500_BST-2023-0343CC43","doi-asserted-by":"publisher","first-page":"12","DOI":"10.1016\/j.gep.2018.01.003","article-title":"Early development of the enteric nervous system visualized by using a new transgenic zebrafish line harboring a regulatory region for choline acetyltransferase a (chata) gene","volume":"28","year":"2018","journal-title":"Gene Expr. Patterns"},{"key":"2024022816503608500_BST-2023-0343CC44","doi-asserted-by":"publisher","first-page":"6941","DOI":"10.1038\/s41598-019-43497-9","article-title":"Immunohistochemical and ultrastructural analysis of the maturing larval zebrafish enteric nervous system reveals the formation of a neuropil pattern","volume":"9","year":"2019","journal-title":"Sci. Rep."},{"key":"2024022816503608500_BST-2023-0343CC45","first-page":"143","volume-title":"The Zebrafish: Cellular and Developmental Biology, Part B","author":"Detrich","year":"2011"},{"key":"2024022816503608500_BST-2023-0343CC46","doi-asserted-by":"publisher","first-page":"1213","DOI":"10.1042\/BST20221375","article-title":"Mechanisms of epithelial growth and development in the zebrafish intestine","volume":"51","year":"2023","journal-title":"Biochem. Soc. Trans."},{"key":"2024022816503608500_BST-2023-0343CC47","doi-asserted-by":"publisher","first-page":"iyac016","DOI":"10.1093\/genetics\/iyac016","article-title":"Zebrafish information network, the knowledgebase for Danio rerio research","volume":"220","year":"2022","journal-title":"Genetics"},{"key":"2024022816503608500_BST-2023-0343CC48","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1002\/dvdy.122","article-title":"Neural crest development: insights from the zebrafish","volume":"249","year":"2020","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC49","doi-asserted-by":"publisher","first-page":"508","DOI":"10.1016\/j.ydbio.2008.02.058","article-title":"Sequential actions of Pax3 and Pax7 drive xanthophore development in zebrafish neural crest","volume":"317","year":"2008","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC50","doi-asserted-by":"publisher","first-page":"1987","DOI":"10.1242\/dev.033209","article-title":"Genetic ablation of neural crest cell diversification","volume":"136","year":"2009","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC51","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1016\/j.ydbio.2011.09.019","article-title":"Tfap2a and Foxd3 regulate early steps in the development of the neural crest progenitor population","volume":"360","year":"2011","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC52","doi-asserted-by":"publisher","first-page":"515","DOI":"10.1242\/dev.127.3.515","article-title":"The zebrafish colourless gene regulates development of non-ectomesenchymal neural crest derivatives","volume":"127","year":"2000","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC53","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1002\/gene.1062","article-title":"A morpholino phenocopy of the colourless mutant","volume":"30","year":"2001","journal-title":"Genesis"},{"key":"2024022816503608500_BST-2023-0343CC54","doi-asserted-by":"publisher","first-page":"3199","DOI":"10.1002\/dvdy.20959","article-title":"The mother superior mutation ablates foxd3 activity in neural crest progenitor cells and depletes neural crest derivatives in zebrafish","volume":"235","year":"2006","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC55","doi-asserted-by":"publisher","first-page":"1060","DOI":"10.1002\/dvdy.21485","article-title":"Zebrafish sip1a and sip1b are essential for normal axial and neural patterning","volume":"237","year":"2008","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC56","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1016\/j.ydbio.2013.01.013","article-title":"Loss of ascl1a prevents secretory cell differentiation within the zebrafish intestinal epithelium resulting in a loss of distal intestinal motility","volume":"376","year":"2013","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC57","doi-asserted-by":"publisher","first-page":"304","DOI":"10.1111\/j.1365-2982.2008.01234.x","article-title":"Analysis of gastrointestinal physiology using a novel intestinal transit assay in zebrafish","volume":"21","year":"2009","journal-title":"Neurogastroenterol. Motil."},{"key":"2024022816503608500_BST-2023-0343CC58","doi-asserted-by":"publisher","first-page":"581","DOI":"10.1016\/j.ajhg.2015.02.014","article-title":"Functional loss of semaphorin 3C and\/or semaphorin 3D and their epistatic interaction with ret are critical to Hirschsprung disease liability","volume":"96","year":"2015","journal-title":"Am. J. Hum. Genet."},{"key":"2024022816503608500_BST-2023-0343CC59","doi-asserted-by":"publisher","first-page":"1421","DOI":"10.1056\/NEJMoa1706594","article-title":"Molecular genetic anatomy and risk profile of Hirschsprung's disease","volume":"380","year":"2019","journal-title":"N. Engl. J. Med."},{"key":"2024022816503608500_BST-2023-0343CC60","doi-asserted-by":"publisher","first-page":"e1009698","DOI":"10.1371\/journal.pgen.1009698","article-title":"Size matters: large copy number losses in Hirschsprung disease patients reveal genes involved in enteric nervous system development","volume":"17","year":"2021","journal-title":"PLoS Genet."},{"key":"2024022816503608500_BST-2023-0343CC61","doi-asserted-by":"publisher","first-page":"523","DOI":"10.3389\/fncel.2019.00523","article-title":"BMP2 is related to Hirschsprung's disease and required for enteric nervous system development","volume":"13","year":"2019","journal-title":"Front. Cell. Neurosci."},{"key":"2024022816503608500_BST-2023-0343CC62","doi-asserted-by":"publisher","first-page":"17","DOI":"10.1016\/j.ydbio.2017.10.021","article-title":"Retinoic acid temporally orchestrates colonization of the gut by vagal neural crest cells","volume":"433","year":"2018","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC63","doi-asserted-by":"publisher","first-page":"34","DOI":"10.1038\/s41593-020-00736-x","article-title":"Diversification of molecularly defined myenteric neuron classes revealed by single-cell RNA sequencing","volume":"24","year":"2021","journal-title":"Nat. Neurosci."},{"key":"2024022816503608500_BST-2023-0343CC64","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1038\/s41467-021-27594-w","article-title":"Lifelong single-cell profiling of cranial neural crest diversification in zebrafish","volume":"13","year":"2022","journal-title":"Nat. Commun."},{"key":"2024022816503608500_BST-2023-0343CC65","doi-asserted-by":"publisher","first-page":"e2212578120","DOI":"10.1073\/pnas.2212578120","article-title":"Post-transcriptional regulation in cranial neural crest cells expands developmental potential","volume":"120","year":"2023","journal-title":"Proc. Natl Acad. Sci. U.S.A."},{"key":"2024022816503608500_BST-2023-0343CC66","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1038\/npp.2012.112","article-title":"DNA methylation and its basic function","volume":"38","year":"2013","journal-title":"Neuropsychopharmacology"},{"key":"2024022816503608500_BST-2023-0343CC67","doi-asserted-by":"publisher","first-page":"1760","DOI":"10.1126\/science.1147939","article-title":"UHRF1 plays a role in maintaining DNA methylation in mammalian cells","volume":"317","year":"2007","journal-title":"Science"},{"key":"2024022816503608500_BST-2023-0343CC68","doi-asserted-by":"publisher","first-page":"473","DOI":"10.1016\/j.ydbio.2019.08.002","article-title":"Epigenetic factors Dnmt1 and Uhrf1 coordinate intestinal development","volume":"455","year":"2019","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC69","doi-asserted-by":"publisher","first-page":"19238","DOI":"10.1073\/pnas.1213825109","article-title":"Chromatin remodeling by the CHD7 protein is impaired by mutations that cause human developmental disorders","volume":"109","year":"2012","journal-title":"Proc. Natl Acad. Sci. U.S.A."},{"key":"2024022816503608500_BST-2023-0343CC70","doi-asserted-by":"publisher","first-page":"e1001023","DOI":"10.1371\/journal.pgen.1001023","article-title":"CHD7 targets active gene enhancer elements to modulate ES cell-specific gene expression","volume":"6","year":"2010","journal-title":"PLoS Genet."},{"key":"2024022816503608500_BST-2023-0343CC71","doi-asserted-by":"publisher","first-page":"3539","DOI":"10.1093\/hmg\/ddw198","article-title":"Rescue of neural crest-derived phenotypes in a zebrafish CHARGE model by Sox10 downregulation","volume":"25","year":"2016","journal-title":"Hum. Mol. Genet."},{"key":"2024022816503608500_BST-2023-0343CC72","doi-asserted-by":"publisher","first-page":"955","DOI":"10.1038\/ng1407","article-title":"Mutations in a new member of the chromodomain gene family cause CHARGE syndrome","volume":"36","year":"2004","journal-title":"Nat. Genet."},{"key":"2024022816503608500_BST-2023-0343CC73","doi-asserted-by":"publisher","first-page":"103661","DOI":"10.1016\/j.ejmg.2019.04.018","article-title":"Chemical screens in a zebrafish model of CHARGE syndrome identifies small molecules that ameliorate disease-like phenotypes in embryo","volume":"63","year":"2020","journal-title":"Eur. J. Med. Genet."},{"key":"2024022816503608500_BST-2023-0343CC74","doi-asserted-by":"publisher","first-page":"2125","DOI":"10.1111\/febs.14473","article-title":"Etiology and functional validation of gastrointestinal motility dysfunction in a zebrafish model of CHARGE syndrome","volume":"285","year":"2018","journal-title":"FEBS J."},{"key":"2024022816503608500_BST-2023-0343CC75","doi-asserted-by":"publisher","first-page":"960149","DOI":"10.3389\/fnins.2022.960149","article-title":"The Polycomb group gene rnf2 is essential for central and enteric neural system development in zebrafish","volume":"16","year":"2022","journal-title":"Front. Neurosci."},{"key":"2024022816503608500_BST-2023-0343CC76","doi-asserted-by":"publisher","first-page":"1020","DOI":"10.1016\/j.molcel.2019.03.024","article-title":"Synergy between variant PRC1 complexes defines polycomb-mediated gene repression","volume":"74","year":"2019","journal-title":"Mol. Cell"},{"key":"2024022816503608500_BST-2023-0343CC77","doi-asserted-by":"publisher","first-page":"48","DOI":"10.1186\/s13059-017-1174-6","article-title":"Whole exome sequencing coupled with unbiased functional analysis reveals new Hirschsprung disease genes","volume":"18","year":"2017","journal-title":"Genome Biol."},{"key":"2024022816503608500_BST-2023-0343CC78","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1016\/j.ydbio.2005.04.023","article-title":"Computational representation of developmental genetic regulatory networks","volume":"283","year":"2005","journal-title":"Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC79","doi-asserted-by":"publisher","first-page":"110311","DOI":"10.1016\/j.celrep.2022.110311","article-title":"A cell atlas of microbe-responsive processes in the zebrafish intestine","volume":"38","year":"2022","journal-title":"Cell Rep."},{"key":"2024022816503608500_BST-2023-0343CC80","doi-asserted-by":"publisher","first-page":"112095","DOI":"10.1016\/j.celrep.2023.112095","article-title":"Cell-type-specific responses to the microbiota across all tissues of the larval zebrafish","volume":"42","year":"2023","journal-title":"Cell Rep."},{"key":"2024022816503608500_BST-2023-0343CC81","doi-asserted-by":"publisher","first-page":"143","DOI":"10.1038\/nn2025","article-title":"CNS-derived glia ensheath peripheral nerves and mediate motor root development","volume":"11","year":"2008","journal-title":"Nat. Neurosci."},{"key":"2024022816503608500_BST-2023-0343CC82","doi-asserted-by":"publisher","first-page":"902","DOI":"10.1053\/j.gastro.2008.11.012","article-title":"Abnormal nuclear pore formation triggers apoptosis in the intestinal epithelium of elys-deficient zebrafish","volume":"136","year":"2009","journal-title":"Gastroenterology"},{"key":"2024022816503608500_BST-2023-0343CC83","doi-asserted-by":"publisher","first-page":"e32899","DOI":"10.1371\/journal.pone.0032899","article-title":"Zebrafish arl6ip1 is required for neural crest development during embryogenesis","volume":"7","year":"2012","journal-title":"PLoS One"},{"key":"2024022816503608500_BST-2023-0343CC84","doi-asserted-by":"publisher","first-page":"1","DOI":"10.3389\/fcell.2021.641152","article-title":"Association analysis of variants of DSCAM and BACE2 with Hirschsprung disease susceptibility in Han Chinese and functional evaluation in zebrafish","volume":"9","year":"2021","journal-title":"Front. Cell Dev. Biol."},{"key":"2024022816503608500_BST-2023-0343CC85","doi-asserted-by":"publisher","first-page":"263","DOI":"10.1016\/j.cell.2014.06.017","article-title":"Disruptive CHD8 mutations define a subtype of autism early in development","volume":"158","year":"2014","journal-title":"Cell"},{"key":"2024022816503608500_BST-2023-0343CC86","doi-asserted-by":"crossref","unstructured":"Davidson, A.E., Straquadine, N.R.W., Cook, S.A., Liu, C.G. and Ganz, J. A rapid F0 CRISPR screen in zebrafish to identify regulators of neuronal development in the enteric nervous system. bioRxiv. 2021.07.17.452230.10.1101\/2021.07.17.452230","DOI":"10.1101\/2021.07.17.452230"},{"key":"2024022816503608500_BST-2023-0343CC87","doi-asserted-by":"publisher","first-page":"e1004946","DOI":"10.1371\/journal.pgen.1004946","article-title":"Fascin1-dependent filopodia are required for directional migration of a subset of neural crest cells","volume":"11","year":"2015","journal-title":"PLoS Genet."},{"key":"2024022816503608500_BST-2023-0343CC88","doi-asserted-by":"publisher","first-page":"118","DOI":"10.1002\/dvdy.21033","article-title":"Genetic screen for mutations affecting development and function of the enteric nervous system","volume":"236","year":"2007","journal-title":"Dev. Dyn."},{"key":"2024022816503608500_BST-2023-0343CC89","doi-asserted-by":"publisher","first-page":"147","DOI":"10.1089\/zeb.2012.0811","article-title":"Characterization of enteric neurons in wild-type and mutant zebrafish using semi-automated cell counting and co-expression analysis","volume":"10","year":"2013","journal-title":"Zebrafish"},{"key":"2024022816503608500_BST-2023-0343CC90","doi-asserted-by":"publisher","first-page":"11316","DOI":"10.1038\/s41598-020-68172-2","article-title":"Her9\/Hes4 is required for retinal photoreceptor development, maintenance, and survival","volume":"10","year":"2020","journal-title":"Sci. Rep."},{"key":"2024022816503608500_BST-2023-0343CC91","doi-asserted-by":"publisher","first-page":"e63218","DOI":"10.1371\/journal.pone.0063218","article-title":"Distinct functional and temporal requirements for zebrafish Hdac1 during neural crest-derived craniofacial and peripheral neuron development","volume":"8","year":"2013","journal-title":"PLoS One"},{"key":"2024022816503608500_BST-2023-0343CC92","doi-asserted-by":"publisher","first-page":"118","DOI":"10.1053\/j.gastro.2018.03.034","article-title":"Identification of variants in RET and IHH pathway members in a large family with history of Hirschsprung disease","volume":"155","year":"2018","journal-title":"Gastroenterology"},{"key":"2024022816503608500_BST-2023-0343CC93","doi-asserted-by":"publisher","first-page":"599","DOI":"10.1242\/dev.012377","article-title":"KBP is essential for axonal structure, outgrowth and maintenance in zebrafish, providing insight into the cellular basis of Goldberg-Shprintzen syndrome","volume":"135","year":"2008","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC94","doi-asserted-by":"publisher","first-page":"345","DOI":"10.1111\/nmo.12732","article-title":"The zebrafish mutant lessen: an experimental model for congenital enteric neuropathies","volume":"28","year":"2016","journal-title":"Neurogastroenterol. Motil."},{"key":"2024022816503608500_BST-2023-0343CC95","doi-asserted-by":"publisher","first-page":"1182","DOI":"10.1016\/j.jpedsurg.2017.01.005","article-title":"Neuregulin 1 is involved in enteric nervous system development in zebrafish","volume":"52","year":"2017","journal-title":"J. Pediatr. Surg."},{"key":"2024022816503608500_BST-2023-0343CC96","doi-asserted-by":"publisher","first-page":"dev180133","DOI":"10.1242\/dev.180133","article-title":"The Paf1 complex and P-TEFb have reciprocal and antagonist roles in maintaining multipotent neural crest progenitors","volume":"146","year":"2019","journal-title":"Development"},{"key":"2024022816503608500_BST-2023-0343CC97","doi-asserted-by":"publisher","first-page":"157","DOI":"10.1016\/j.mod.2004.10.009","article-title":"Intestinal growth and differentiation in zebrafish","volume":"122","year":"2005","journal-title":"Mech. Dev."},{"key":"2024022816503608500_BST-2023-0343CC98","doi-asserted-by":"publisher","first-page":"750","DOI":"10.1016\/j.devcel.2010.03.009","article-title":"Phosphatase-dependent and -independent functions of Shp2 in neural crest cells underlie LEOPARD syndrome pathogenesis","volume":"18","year":"2010","journal-title":"Dev. Cell"},{"key":"2024022816503608500_BST-2023-0343CC99","doi-asserted-by":"publisher","first-page":"771","DOI":"10.1053\/j.gastro.2014.12.034","article-title":"Mutations in RAD21 disrupt regulation of APOB in patients with chronic intestinal pseudo-obstruction","volume":"148","year":"2015","journal-title":"Gastroenterology"},{"key":"2024022816503608500_BST-2023-0343CC100","doi-asserted-by":"publisher","first-page":"2131","DOI":"10.1101\/gad.1561507","article-title":"A Rap GTPase interactor, RADIL, mediates migration of neural crest precursors","volume":"21","year":"2007","journal-title":"Genes Dev."},{"key":"2024022816503608500_BST-2023-0343CC101","doi-asserted-by":"publisher","first-page":"15667","DOI":"10.3390\/ijms232415667","article-title":"A new transgenic tool to study the ret signaling pathway in the enteric nervous system","volume":"23","year":"2022","journal-title":"Int. J. Mol. Sci."},{"key":"2024022816503608500_BST-2023-0343CC102","doi-asserted-by":"publisher","first-page":"e2000689","DOI":"10.1371\/journal.pbio.2000689","article-title":"The enteric nervous system promotes intestinal health by constraining microbiota composition","volume":"15","year":"2017","journal-title":"PLoS Biol."},{"key":"2024022816503608500_BST-2023-0343CC103","doi-asserted-by":"publisher","first-page":"6714","DOI":"10.1073\/pnas.0707057105","article-title":"Inhibition of neural crest migration underlies craniofacial dysmorphology and Hirschsprung's disease in Bardet-Biedl syndrome","volume":"105","year":"2008","journal-title":"Proc. Natl Acad. Sci. U.S.A."},{"key":"2024022816503608500_BST-2023-0343CC104","doi-asserted-by":"publisher","first-page":"1","DOI":"10.3389\/fcell.2022.901824","article-title":"TFAP2B haploinsufficiency impacts gastrointestinal function and leads to pediatric intestinal pseudo-obstruction","volume":"10","year":"2022","journal-title":"Front. Cell Dev. Biol."}],"container-title":["Biochemical Society Transactions"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/portlandpress.com\/biochemsoctrans\/article-pdf\/52\/1\/177\/954662\/bst-2023-0343c.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/portlandpress.com\/biochemsoctrans\/article-pdf\/52\/1\/177\/954662\/bst-2023-0343c.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,28]],"date-time":"2024-02-28T11:50:52Z","timestamp":1709121052000},"score":1,"resource":{"primary":{"URL":"https:\/\/portlandpress.com\/biochemsoctrans\/article\/52\/1\/177\/233942\/Genetic-regulation-of-enteric-nervous-system"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,4]]},"references-count":104,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,1,4]]},"published-print":{"date-parts":[[2024,2,28]]}},"URL":"https:\/\/doi.org\/10.1042\/bst20230343","relation":{},"ISSN":["0300-5127","1470-8752"],"issn-type":[{"value":"0300-5127","type":"print"},{"value":"1470-8752","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,1,4]]}}}