{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T03:28:15Z","timestamp":1792812495025,"version":"4.5.0"},"reference-count":121,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T00:00:00Z","timestamp":1677628800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T00:00:00Z","timestamp":1677628800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,2,10]],"date-time":"2023-02-10T00:00:00Z","timestamp":1675987200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["cell.com","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Cell Stem Cell"],"published-print":{"date-parts":[[2023,3]]},"DOI":"10.1016\/j.stem.2023.02.003","type":"journal-article","created":{"date-parts":[[2023,3,2]],"date-time":"2023-03-02T10:40:43Z","timestamp":1677753643000},"page":"264-282.e9","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":54,"title":["hPSC-derived sacral neural crest enables rescue in a severe model of Hirschsprung\u2019s disease"],"prefix":"10.1016","volume":"30","author":[{"given":"Yujie","family":"Fan","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Hackland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Arianna","family":"Baggiolini","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lin Y.","family":"Hung","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huiyong","family":"Zhao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Zumbo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Polina","family":"Oberst","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew P.","family":"Minotti","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emiliano","family":"Hergenreder","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Najjar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zixing","family":"Huang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nelly M.","family":"Cruz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aaron","family":"Zhong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mega","family":"Sidharta","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ting","family":"Zhou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisa","family":"de Stanchina","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Doron","family":"Betel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard M.","family":"White","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael","family":"Gershon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kara Gross","family":"Margolis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0741-7987","authenticated-orcid":false,"given":"Lorenz","family":"Studer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.stem.2023.02.003_bib1","series-title":"The Second Brain: a Groundbreaking New Understanding of Nervous Disorders of the Stomach and Intestine","author":"Gershon","year":"1999"},{"key":"10.1016\/j.stem.2023.02.003_bib2","doi-asserted-by":"crossref","first-page":"261","DOI":"10.1111\/j.1469-1809.1963.tb01983.x","article-title":"A family study of Hirschsprung's disease","volume":"26","author":"Bodian","year":"1963","journal-title":"Ann. Hum. Genet."},{"key":"10.1016\/j.stem.2023.02.003_bib3","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1001\/archinte.1948.00220250085005","article-title":"Myenteric plexus in congenital megacolon: study of Eleven Cases","volume":"82","author":"Whitehouse","year":"1948","journal-title":"Arch. Intern. Med. (Chic)"},{"key":"10.1016\/j.stem.2023.02.003_bib4","doi-asserted-by":"crossref","first-page":"409","DOI":"10.1016\/S0046-8177(74)80021-3","article-title":"The neurocristopathies: a unifying concept of disease arising in neural crest maldevelopment","volume":"5","author":"Bolande","year":"1974","journal-title":"Hum. Pathol."},{"key":"10.1016\/j.stem.2023.02.003_bib5","doi-asserted-by":"crossref","first-page":"466","DOI":"10.1038\/nrn2137","article-title":"Enteric nervous system development and Hirschsprung's disease: advances in genetic and stem cell studies","volume":"8","author":"Heanue","year":"2007","journal-title":"Nat. Rev. Neurosci."},{"key":"10.1016\/j.stem.2023.02.003_bib6","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1016\/S0022-3468(86)80847-8","article-title":"Total colonic aganglionosis (with or without ileal involvement): a review of 27 cases","volume":"21","author":"N-F\u00e9k\u00e9t\u00e9","year":"1986","journal-title":"J.\u00a0Pediatr. Surg."},{"key":"10.1016\/j.stem.2023.02.003_bib7","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1016\/S0959-437X(99)80048-3","article-title":"Development of the mammalian enteric nervous system","volume":"9","author":"Taraviras","year":"1999","journal-title":"Curr. Opin. Genet. Dev."},{"key":"10.1016\/j.stem.2023.02.003_bib8","doi-asserted-by":"crossref","first-page":"1267","DOI":"10.1016\/0092-8674(94)90017-5","article-title":"Targeted and natural (piebald-lethal) mutations of endothelin-B receptor gene produce megacolon associated with spotted coat color in mice","volume":"79","author":"Hosoda","year":"1994","journal-title":"Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib9","doi-asserted-by":"crossref","first-page":"229","DOI":"10.1016\/j.ydbio.2016.04.001","article-title":"White paper on guidelines concerning enteric nervous system stem cell therapy for enteric neuropathies","volume":"417","author":"Burns","year":"2016","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib10","doi-asserted-by":"crossref","first-page":"517","DOI":"10.1038\/nrgastro.2016.107","article-title":"The bowel and beyond: the enteric nervous system in neurological disorders","volume":"13","author":"Rao","year":"2016","journal-title":"Nat. Rev. Gastroenterol. Hepatol."},{"key":"10.1016\/j.stem.2023.02.003_bib11","series-title":"Neuroanatomy and Pathology of Sporadic Parkinson's Disease (Advances in Anatomy, Embryology and Cell Biology; 201)","author":"Braak","year":"2009"},{"key":"10.1016\/j.stem.2023.02.003_bib12","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1016\/S0197-4580(02)00065-9","article-title":"Staging of brain pathology related to sporadic Parkinson\u2019s disease","volume":"24","author":"Braak","year":"2003","journal-title":"Neurobiol. Aging"},{"key":"10.1016\/j.stem.2023.02.003_bib13","doi-asserted-by":"crossref","first-page":"552","DOI":"10.1038\/s41583-018-0041-0","article-title":"Enteric nervous system development: what could possibly go wrong?","volume":"19","author":"Rao","year":"2018","journal-title":"Nat. Rev. Neurosci."},{"key":"10.1016\/j.stem.2023.02.003_bib14","doi-asserted-by":"crossref","first-page":"G1","DOI":"10.1152\/ajpgi.00452.2012","article-title":"Enteric nervous system development: migration, differentiation, and disease","volume":"305","author":"Lake","year":"2013","journal-title":"Am. J. Physiol. Gastrointest. Liver Physiol."},{"key":"10.1016\/j.stem.2023.02.003_bib15","doi-asserted-by":"crossref","first-page":"146","DOI":"10.1006\/dbio.2000.9886","article-title":"Colonization of the murine hindgut by sacral crest-derived neural precursors: experimental support for an evolutionarily conserved model","volume":"227","author":"Kapur","year":"2000","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib16","doi-asserted-by":"crossref","first-page":"715","DOI":"10.1038\/nbt.2249","article-title":"Combined small-molecule inhibition accelerates developmental timing and converts human pluripotent stem cells into nociceptors","volume":"30","author":"Chambers","year":"2012","journal-title":"Nat. Biotechnol."},{"key":"10.1016\/j.stem.2023.02.003_bib17","doi-asserted-by":"crossref","first-page":"19240","DOI":"10.1073\/pnas.1113746108","article-title":"Wnt signaling and a Smad pathway blockade direct the differentiation of human pluripotent stem cells to multipotent neural crest cells","volume":"108","author":"Menendez","year":"2011","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"10.1016\/j.stem.2023.02.003_bib18","doi-asserted-by":"crossref","first-page":"1043","DOI":"10.1016\/j.stemcr.2017.08.008","article-title":"Top-down inhibition of BMP signaling enables robust induction of hPSCs into neural crest in fully defined, xeno-free conditions","volume":"9","author":"Hackland","year":"2017","journal-title":"Stem Cell Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib19","doi-asserted-by":"crossref","first-page":"399","DOI":"10.1016\/j.stem.2017.08.015","article-title":"A modular platform for differentiation of human PSCs into all major ectodermal lineages","volume":"21","author":"Tchieu","year":"2017","journal-title":"Cell Stem Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib20","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1038\/nature16951","article-title":"Deriving human ENS lineages for cell therapy and drug discovery in Hirschsprung disease","volume":"531","author":"Fattahi","year":"2016","journal-title":"Nature"},{"key":"10.1016\/j.stem.2023.02.003_bib21","doi-asserted-by":"crossref","first-page":"19727","DOI":"10.1038\/srep19727","article-title":"Generating trunk neural crest from human pluripotent stem cells","volume":"6","author":"Huang","year":"2016","journal-title":"Sci. Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib22","doi-asserted-by":"crossref","first-page":"1140","DOI":"10.1016\/j.celrep.2013.03.025","article-title":"Modeling neural crest induction, melanocyte specification, and disease-related pigmentation defects in hESCs and patient-specific iPSCs","volume":"3","author":"Mica","year":"2013","journal-title":"Cell Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib23","doi-asserted-by":"crossref","first-page":"e35786","DOI":"10.7554\/eLife.35786","article-title":"Human axial progenitors generate trunk neural crest cells in\u00a0vitro","volume":"7","author":"Frith","year":"2018","journal-title":"eLife"},{"key":"10.1016\/j.stem.2023.02.003_bib24","article-title":"Neuromesodermal progenitor origin of trunk neural crest in\u00a0vivo","author":"Martyna Lukoseviciute","year":"2021","journal-title":"Preprint at bioRxiv"},{"key":"10.1016\/j.stem.2023.02.003_bib25","doi-asserted-by":"crossref","first-page":"2864","DOI":"10.1242\/dev.119768","article-title":"Neuromesodermal progenitors and the making of the spinal cord","volume":"142","author":"Henrique","year":"2015","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib26","doi-asserted-by":"crossref","first-page":"e1001937","DOI":"10.1371\/journal.pbio.1001937","article-title":"In\u00a0vitro generation of neuromesodermal progenitors reveals distinct roles for wnt signalling in the specification of spinal cord and paraxial mesoderm identity","volume":"12","author":"Gouti","year":"2014","journal-title":"PLoS Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib27","doi-asserted-by":"crossref","first-page":"632","DOI":"10.1016\/j.stemcr.2015.02.018","article-title":"Deterministic HOX patterning in human pluripotent stem cell-derived neuroectoderm","volume":"4","author":"Lippmann","year":"2015","journal-title":"Stem Cell Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib28","doi-asserted-by":"crossref","first-page":"100","DOI":"10.12688\/f1000research.6345.2","article-title":"Assessing the bipotency of in\u00a0vitro-derived neuromesodermal progenitors","volume":"4","author":"Tsakiridis","year":"2015","journal-title":"F1000Res"},{"key":"10.1016\/j.stem.2023.02.003_bib29","doi-asserted-by":"crossref","first-page":"920","DOI":"10.1016\/j.stemcr.2019.04.015","article-title":"FGF modulates the axial identity of trunk hPSC-derived neural crest but not the cranial-trunk decision","volume":"12","author":"Hackland","year":"2019","journal-title":"Stem Cell Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib30","doi-asserted-by":"crossref","first-page":"1209","DOI":"10.1242\/dev.101014","article-title":"Distinct Wnt-driven primitive streak-like populations reflect in\u00a0vivo lineage precursors","volume":"141","author":"Tsakiridis","year":"2014","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib31","doi-asserted-by":"crossref","first-page":"4243","DOI":"10.1242\/dev.112979","article-title":"Wnt\/beta-catenin and FGF signalling direct the specification and maintenance of a neuromesodermal axial progenitor in ensembles of mouse embryonic stem cells","volume":"141","author":"Turner","year":"2014","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib32","doi-asserted-by":"crossref","first-page":"336","DOI":"10.1053\/j.sempedsurg.2012.07.008","article-title":"Long-term outcomes of Hirschsprung's disease","volume":"21","author":"Rintala","year":"2012","journal-title":"Semin. Pediatr. Surg."},{"key":"10.1016\/j.stem.2023.02.003_bib33","doi-asserted-by":"crossref","first-page":"622","DOI":"10.1097\/SLA.0000000000001695","article-title":"Bowel function and quality of life after transanal endorectal pull-through for Hirschsprung disease: controlled outcomes up to adulthood","volume":"265","author":"Neuvonen","year":"2017","journal-title":"Ann. Surg."},{"key":"10.1016\/j.stem.2023.02.003_bib34","doi-asserted-by":"crossref","first-page":"S170","DOI":"10.1016\/j.ydbio.2018.07.026","article-title":"The molecular basis of neural crest axial identity","volume":"444","author":"Rothstein","year":"2018","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib35","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1016\/S0925-4773(98)00205-6","article-title":"Expression of growth\/differentiation factor 11, a new member of the BMP\/TGFbeta superfamily during mouse embryogenesis","volume":"80","author":"Nakashima","year":"1999","journal-title":"Mech. Dev."},{"key":"10.1016\/j.stem.2023.02.003_bib36","doi-asserted-by":"crossref","first-page":"260","DOI":"10.1038\/10320","article-title":"Regulation of anterior\/posterior patterning of the axial skeleton by growth\/differentiation factor 11","volume":"22","author":"McPherron","year":"1999","journal-title":"Nat. Genet."},{"key":"10.1016\/j.stem.2023.02.003_bib37","doi-asserted-by":"crossref","first-page":"2865","DOI":"10.1242\/dev.02478","article-title":"The function of growth\/differentiation factor 11 (Gdf11) in rostrocaudal patterning of the developing spinal cord","volume":"133","author":"Liu","year":"2006","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib38","doi-asserted-by":"crossref","first-page":"23360","DOI":"10.1002\/jcp.28904","article-title":"Growth differentiation factor 11 locally controls anterior-posterior patterning of the axial skeleton","volume":"234","author":"Suh","year":"2019","journal-title":"J.\u00a0Cell. Physiol."},{"key":"10.1016\/j.stem.2023.02.003_bib39","doi-asserted-by":"crossref","first-page":"451","DOI":"10.1016\/j.devcel.2013.05.009","article-title":"Switching axial progenitors from producing trunk to tail tissues in vertebrate embryos","volume":"25","author":"Jurberg","year":"2013","journal-title":"Dev. Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib40","doi-asserted-by":"crossref","first-page":"1465","DOI":"10.1101\/gad.479408","article-title":"VACTERL\/caudal regression\/Currarino syndrome-like malformations in mice with mutation in the proprotein convertase Pcsk5","volume":"22","author":"Szumska","year":"2008","journal-title":"Genes Dev."},{"key":"10.1016\/j.stem.2023.02.003_bib41","doi-asserted-by":"crossref","first-page":"15764","DOI":"10.1038\/s41598-018-33592-8","article-title":"EzColocalization: an ImageJ plugin for visualizing and measuring colocalization in cells and organisms","volume":"8","author":"Stauffer","year":"2018","journal-title":"Sci. Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib42","first-page":"51609","article-title":"Feeder-free derivation of neural crest progenitor cells from human pluripotent stem cells","volume":"87","author":"Zeltner","year":"2014","journal-title":"J. Vis. Exp."},{"key":"10.1016\/j.stem.2023.02.003_bib43","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1242\/dev.121.2.333","article-title":"Hox genes and the evolution of vertebrate axial morphology","volume":"121","author":"Burke","year":"1995","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib44","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1242\/dev.101.1.1","article-title":"The molecular basis for metameric pattern in the Drosophila embryo","volume":"101","author":"Akam","year":"1987","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib45","doi-asserted-by":"crossref","first-page":"95","DOI":"10.1098\/rspb.1979.0093","article-title":"Neural tube defects in curly-tail mice. II. Effect of maternal administration of vitamin A","volume":"206","author":"Seller","year":"1979","journal-title":"Proc. R. Soc. Lond. B Biol. Sci."},{"key":"10.1016\/j.stem.2023.02.003_bib46","doi-asserted-by":"crossref","first-page":"369","DOI":"10.1016\/j.stem.2010.02.014","article-title":"An Oct4-centered protein interaction network in embryonic stem cells","volume":"6","author":"van den Berg","year":"2010","journal-title":"Cell Stem Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib47","doi-asserted-by":"crossref","first-page":"40852","DOI":"10.1074\/jbc.M110.103812","article-title":"Grainyhead-like 2 enhances the human telomerase reverse transcriptase gene expression by inhibiting DNA methylation at the 5\u2032-CpG island in normal human keratinocytes","volume":"285","author":"Chen","year":"2010","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.stem.2023.02.003_bib48","doi-asserted-by":"crossref","first-page":"e1002829","DOI":"10.1371\/journal.pgen.1002829","article-title":"GRHL3\/GET1 and trithorax group members collaborate to activate the epidermal progenitor differentiation program","volume":"8","author":"Hopkin","year":"2012","journal-title":"PLoS Genet."},{"key":"10.1016\/j.stem.2023.02.003_bib49","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1016\/j.ydbio.2010.08.022","article-title":"Growth differentiation factor 11 signaling controls retinoic acid activity for axial vertebral development","volume":"347","author":"Lee","year":"2010","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib50","doi-asserted-by":"crossref","first-page":"7158","DOI":"10.1128\/MCB.19.10.7158","article-title":"Physical and functional interactions between cellular retinoic acid binding protein II and the retinoic acid-dependent nuclear complex","volume":"19","author":"Delva","year":"1999","journal-title":"Mol. Cell. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib51","doi-asserted-by":"crossref","first-page":"16","DOI":"10.1186\/s12976-018-0088-7","article-title":"Identification of influential proteins in the classical retinoic acid signaling pathway","volume":"15","author":"Ghaffari","year":"2018","journal-title":"Theor. Biol. Med. Model."},{"key":"10.1016\/j.stem.2023.02.003_bib52","doi-asserted-by":"crossref","first-page":"489","DOI":"10.1083\/jcb.201011077","article-title":"FGF and retinoic acid activity gradients control the timing of neural crest cell emigration in the trunk","volume":"194","author":"Mart\u00ednez-Morales","year":"2011","journal-title":"J.\u00a0Cell Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib53","doi-asserted-by":"crossref","first-page":"991","DOI":"10.1242\/dev.119.4.991","article-title":"Tail formation as a continuation of gastrulation: the multiple cell populations of the Xenopus tailbud derive from the late blastopore lip","volume":"119","author":"Gont","year":"1993","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib54","doi-asserted-by":"crossref","first-page":"365","DOI":"10.1016\/j.devcel.2009.08.002","article-title":"Redefining the progression of lineage segregations during mammalian embryogenesis by clonal analysis","volume":"17","author":"Tzouanacou","year":"2009","journal-title":"Dev. Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib55","doi-asserted-by":"crossref","first-page":"3165","DOI":"10.1016\/j.celrep.2016.11.069","article-title":"Cdx and T brachyury co-activate growth signaling in the embryonic axial progenitor niche","volume":"17","author":"Amin","year":"2016","journal-title":"Cell Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib56","doi-asserted-by":"crossref","first-page":"4855","DOI":"10.1242\/dev.129.20.4855","article-title":"Axial progenitors with extensive potency are localised to the mouse chordoneural hinge","volume":"129","author":"Cambray","year":"2002","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib57","series-title":"The Neural Crest","author":"Le Douarin","year":"1999"},{"key":"10.1016\/j.stem.2023.02.003_bib58","series-title":"The Neural Crest: Including a Facsimile Reprint of the Neural Crest by Sven H\u00f6rstadius","author":"Hall","year":"1988"},{"key":"10.1016\/j.stem.2023.02.003_bib59","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1006\/dbio.1999.9592","article-title":"Sacral neural crest cells colonise aganglionic hindgut in\u00a0vivo but fail to compensate for lack of enteric ganglia","volume":"219","author":"Burns","year":"2000","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib60","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1016\/j.ydbio.2005.08.040","article-title":"The pattern of neural crest advance in the cecum and colon","volume":"287","author":"Druckenbrod","year":"2005","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib61","doi-asserted-by":"crossref","first-page":"514","DOI":"10.1002\/cne.23423","article-title":"Birthdating of myenteric neuron subtypes in the small intestine of the mouse","volume":"522","author":"Bergner","year":"2014","journal-title":"J.\u00a0Comp. Neurol."},{"key":"10.1016\/j.stem.2023.02.003_bib62","doi-asserted-by":"crossref","first-page":"474","DOI":"10.1002\/cne.21770","article-title":"Bone morphogenetic protein regulation of enteric neuronal phenotypic diversity: relationship to timing of cell cycle exit","volume":"509","author":"Chalazonitis","year":"2008","journal-title":"J.\u00a0Comp. Neurol."},{"key":"10.1016\/j.stem.2023.02.003_bib63","doi-asserted-by":"crossref","first-page":"789","DOI":"10.1002\/cne.903140411","article-title":"Time of origin of neurons in the murine enteric nervous system: sequence in relation to phenotype","volume":"314","author":"Pham","year":"1991","journal-title":"J.\u00a0Comp. Neurol."},{"key":"10.1016\/j.stem.2023.02.003_bib64","doi-asserted-by":"crossref","first-page":"95","DOI":"10.1016\/j.stem.2016.05.002","article-title":"Functional coupling with cardiac muscle promotes maturation of hPSC-derived sympathetic neurons","volume":"19","author":"Oh","year":"2016","journal-title":"Cell Stem Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib65","doi-asserted-by":"crossref","first-page":"eabc1048","DOI":"10.1126\/science.abc1048","article-title":"Developmental chromatin programs determine oncogenic competence in melanoma","volume":"373","author":"Baggiolini","year":"2021","journal-title":"Science"},{"key":"10.1016\/j.stem.2023.02.003_bib66","article-title":"Anatomic position determines oncogenic specificity in melanoma","author":"Weiss","year":"2021","journal-title":"Preprint at bioRxiv"},{"key":"10.1016\/j.stem.2023.02.003_bib67","doi-asserted-by":"crossref","first-page":"4335","DOI":"10.1242\/dev.125.21.4335","article-title":"The sacral neural crest contributes neurons and glia to the post-umbilical gut: spatiotemporal analysis of the development of the enteric nervous system","volume":"125","author":"Burns","year":"1998","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib68","first-page":"31","article-title":"The migration of neural crest cells to the wall of the digestive tract in avian embryo","volume":"30","author":"Le Douarin","year":"1973","journal-title":"J.\u00a0Embryol. Exp. Morphol."},{"key":"10.1016\/j.stem.2023.02.003_bib69","doi-asserted-by":"crossref","first-page":"179","DOI":"10.1053\/j.gastro.2021.08.029","article-title":"Direct interaction of Sox10 with Cadherin-19 mediates early sacral neural crest cell migration: implications for enteric nervous system development defects","volume":"162","author":"Huang","year":"2022","journal-title":"Gastroenterology"},{"key":"10.1016\/j.stem.2023.02.003_bib70","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1387\/ijdb.041810rf","article-title":"Cadherin-mediated cell-cell adhesion and tissue segregation in relation to malignancy","volume":"48","author":"Foty","year":"2004","journal-title":"Int. J. Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib71","doi-asserted-by":"crossref","first-page":"784039","DOI":"10.3389\/fcell.2021.784039","article-title":"Interplay of Eph-Ephrin signalling and cadherin function in cell segregation and boundary formation","volume":"9","author":"Wilkinson","year":"2021","journal-title":"Front. Cell Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib72","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1002\/dvdy.20200","article-title":"Ephrin signaling in\u00a0vivo: look both ways","volume":"232","author":"Davy","year":"2005","journal-title":"Dev. Dyn."},{"key":"10.1016\/j.stem.2023.02.003_bib73","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1016\/j.tcb.2007.03.004","article-title":"Bidirectional Eph-ephrin signaling during axon guidance","volume":"17","author":"Egea","year":"2007","journal-title":"Trends Cell Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib74","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1038\/nrm856","article-title":"Mechanisms and functions of Eph and ephrin signalling","volume":"3","author":"Kullander","year":"2002","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib75","doi-asserted-by":"crossref","first-page":"e1000597","DOI":"10.1371\/journal.pbio.1000597","article-title":"EphrinB\/EphB signaling controls embryonic germ layer separation by contact-induced cell detachment","volume":"9","author":"Rohani","year":"2011","journal-title":"PLoS Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib76","doi-asserted-by":"crossref","first-page":"37","DOI":"10.1007\/s00441-009-0852-4","article-title":"ChAT and NOS in human myenteric neurons: co-existence and co-absence","volume":"338","author":"Beck","year":"2009","journal-title":"Cell Tissue Res."},{"key":"10.1016\/j.stem.2023.02.003_bib77","doi-asserted-by":"crossref","first-page":"162","DOI":"10.1016\/0012-1606(74)90291-7","article-title":"Experimental analysis of the migration and differentiation of neuroblasts of the autonomic nervous system and of neurectodermal mesenchymal derivatives, using a biological cell marking technique","volume":"41","author":"Le Douarin","year":"1974","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib78","first-page":"125","article-title":"Mesenchymal derivatives of the neural crest: analysis of chimaeric quail and chick embryos","volume":"34","author":"Le Li\u00e8vre","year":"1975","journal-title":"J.\u00a0Embryol. Exp. Morphol."},{"key":"10.1016\/j.stem.2023.02.003_bib79","doi-asserted-by":"crossref","first-page":"4637","DOI":"10.1242\/dev.01350","article-title":"Neural crest cell plasticity and its limits","volume":"131","author":"Le Douarin","year":"2004","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib80","series-title":"The Neural Crest","author":"Le Douarin","year":"1999"},{"key":"10.1016\/j.stem.2023.02.003_bib81","doi-asserted-by":"crossref","first-page":"dev193193","DOI":"10.1242\/dev.193193","article-title":"Neural crest lineage analysis: from past to future trajectory","volume":"147","author":"Tang","year":"2020","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib82","doi-asserted-by":"crossref","first-page":"dev193888","DOI":"10.1242\/dev.193888","article-title":"From head to tail: regionalization of the neural crest","volume":"147","author":"Rocha","year":"2020","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib83","doi-asserted-by":"crossref","first-page":"4567","DOI":"10.1242\/dev.00673","article-title":"Dissimilar regulation of cell differentiation in mesencephalic (cranial) and sacral (trunk) neural crest cells in\u00a0vitro","volume":"130","author":"Abzhanov","year":"2003","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib84","doi-asserted-by":"crossref","first-page":"1061","DOI":"10.1242\/dev.129.4.1061","article-title":"Interactions between Hox-negative cephalic neural crest cells and the foregut endoderm in patterning the facial skeleton in the vertebrate head","volume":"129","author":"Couly","year":"2002","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib85","doi-asserted-by":"crossref","first-page":"1979","DOI":"10.1242\/dev.01106","article-title":"Graded potential of neural crest to form cornea, sensory neurons and cartilage along the rostrocaudal axis","volume":"131","author":"Lwigale","year":"2004","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib86","doi-asserted-by":"crossref","first-page":"39","DOI":"10.3389\/fnmol.2019.00039","article-title":"Human pluripotent stem cell-derived neural crest cells for tissue regeneration and disease modeling","volume":"12","author":"Srinivasan","year":"2019","journal-title":"Front. Mol. Neurosci."},{"key":"10.1016\/j.stem.2023.02.003_bib87","doi-asserted-by":"crossref","first-page":"356","DOI":"10.1016\/j.ydbio.2017.04.011","article-title":"Migration pathways of sacral neural crest during development of lower urogenital tract innervation","volume":"429","author":"Wiese","year":"2017","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib88","doi-asserted-by":"crossref","first-page":"3247","DOI":"10.1242\/dev.023176","article-title":"Wnt and FGF signals interact to coordinate growth with cell fate specification during limb development","volume":"135","author":"ten Berge","year":"2008","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib89","doi-asserted-by":"crossref","first-page":"383","DOI":"10.1016\/j.devcel.2018.12.004","article-title":"Tail bud progenitor activity relies on a network comprising Gdf11, Lin28, and Hox13 genes","volume":"48","author":"Aires","year":"2019","journal-title":"Dev. Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib90","doi-asserted-by":"crossref","first-page":"e53608","DOI":"10.7554\/eLife.53608","article-title":"Lin28a\/let-7 pathway modulates the Hox code via Polycomb regulation during axial patterning in vertebrates","volume":"9","author":"Sato","year":"2020","journal-title":"eLife"},{"key":"10.1016\/j.stem.2023.02.003_bib91","doi-asserted-by":"crossref","first-page":"1759","DOI":"10.1002\/stem.1991","article-title":"Multipotent caudal neural progenitors derived from human pluripotent stem cells that give rise to lineages of the central and peripheral nervous system","volume":"33","author":"Denham","year":"2015","journal-title":"Stem Cells"},{"key":"10.1016\/j.stem.2023.02.003_bib92","series-title":"Die Embryonalen Keimbl\u00e4tter und die Gewebe","author":"von K\u00f6lliker","year":"1884"},{"key":"10.1016\/j.stem.2023.02.003_bib93","doi-asserted-by":"crossref","first-page":"869","DOI":"10.1016\/S0960-9822(00)00601-1","article-title":"A region of the vertebrate neural plate in which neighbouring cells can adopt neural or epidermal fates","volume":"10","author":"Brown","year":"2000","journal-title":"Curr. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib94","doi-asserted-by":"crossref","first-page":"568","DOI":"10.1038\/nature04838","article-title":"Collinear activation of Hoxb genes during gastrulation is linked to mesoderm cell ingression","volume":"442","author":"Iimura","year":"2006","journal-title":"Nature"},{"key":"10.1016\/j.stem.2023.02.003_bib95","doi-asserted-by":"crossref","first-page":"2829","DOI":"10.1242\/dev.02877","article-title":"Two distinct sources for a population of maturing axial progenitors","volume":"134","author":"Cambray","year":"2007","journal-title":"Development"},{"key":"10.1016\/j.stem.2023.02.003_bib96","doi-asserted-by":"crossref","first-page":"e1001415","DOI":"10.1371\/journal.pbio.1001415","article-title":"Loss of FGF-dependent mesoderm identity and rise of endogenous retinoid signalling determine cessation of body axis elongation","volume":"10","author":"Olivera-Martinez","year":"2012","journal-title":"PLoS Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib97","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.ydbio.2018.11.017","article-title":"The enteric neural crest progressively loses capacity to form enteric nervous system","volume":"446","author":"Zhang","year":"2019","journal-title":"Dev. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib119","doi-asserted-by":"crossref","first-page":"1145","DOI":"10.1126\/science.282.5391.1145","article-title":"Embryonic stem cell lines derived from human blastocysts","volume":"282","author":"Thomson","year":"1998","journal-title":"Science"},{"key":"10.1016\/j.stem.2023.02.003_bib120","doi-asserted-by":"crossref","first-page":"1353","DOI":"10.1056\/NEJMsr040330","article-title":"Derivation of embryonic stem-cell lines from human blastocysts","volume":"350","author":"Cowan","year":"2004","journal-title":"N. Engl. J. Med."},{"key":"10.1016\/j.stem.2023.02.003_bib121","doi-asserted-by":"crossref","first-page":"803","DOI":"10.1038\/nbt1318","article-title":"Characterization of human embryonic stem cell lines by the International Stem Cell Initiative","volume":"25","author":"Adewumi","year":"2007","journal-title":"Nat. Biotechnol."},{"key":"10.1016\/j.stem.2023.02.003_bib98","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1016\/j.stem.2019.05.001","article-title":"Lipid deprivation induces a stable, na\u00efve-to-primed intermediate state of pluripotency in human PSCs","volume":"25","author":"Cornacchia","year":"2019","journal-title":"Cell Stem Cell"},{"key":"10.1016\/j.stem.2023.02.003_bib99","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: an open-source platform for biological-image analysis","volume":"9","author":"Schindelin","year":"2012","journal-title":"Nat. Methods"},{"key":"10.1016\/j.stem.2023.02.003_bib100","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1186\/s13059-014-0550-8","article-title":"Moderated estimation of fold change and dispersion for RNA-seq data with DESeq2","volume":"15","author":"Love","year":"2014","journal-title":"Genome Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib101","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1038\/s41587-019-0085-3","article-title":"Specification of positional identity in forebrain organoids","volume":"37","author":"Cederquist","year":"2019","journal-title":"Nat. Biotechnol."},{"key":"10.1016\/j.stem.2023.02.003_bib102","doi-asserted-by":"crossref","first-page":"807","DOI":"10.1002\/eji.201444737","article-title":"Enterocolitis causes profound lymphoid depletion in endothelin receptor B-and endothelin 3-null mouse models of Hirschsprung-associated enterocolitis","volume":"45","author":"Frykman","year":"2015","journal-title":"Eur. J. Immunol."},{"key":"10.1016\/j.stem.2023.02.003_bib103","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1093\/bioinformatics\/bts635","article-title":"STAR: ultrafast universal RNA-seq aligner","volume":"29","author":"Dobin","year":"2013","journal-title":"Bioinformatics"},{"key":"10.1016\/j.stem.2023.02.003_bib104","doi-asserted-by":"crossref","first-page":"1185","DOI":"10.1038\/nmeth.2722","article-title":"Systematic evaluation of spliced alignment programs for RNA-seq data","volume":"10","author":"Engstr\u00f6m","year":"2013","journal-title":"Nat. Methods"},{"key":"10.1016\/j.stem.2023.02.003_bib105","doi-asserted-by":"crossref","first-page":"1213","DOI":"10.1038\/nmeth.2688","article-title":"Transposition of native chromatin for fast and sensitive epigenomic profiling of open chromatin, DNA-binding proteins and nucleosome position","volume":"10","author":"Buenrostro","year":"2013","journal-title":"Nat. Methods"},{"key":"10.1016\/j.stem.2023.02.003_bib106","doi-asserted-by":"crossref","first-page":"1754","DOI":"10.1093\/bioinformatics\/btp324","article-title":"Fast and accurate short read alignment with Burrows\u2013Wheeler transform","volume":"25","author":"Li","year":"2009","journal-title":"Bioinformatics"},{"key":"10.1016\/j.stem.2023.02.003_bib107","doi-asserted-by":"crossref","first-page":"2078","DOI":"10.1093\/bioinformatics\/btp352","article-title":"The Sequence Alignment\/Map format and SAMtools","volume":"25","author":"Li","year":"2009","journal-title":"Bioinformatics Oxf. Engl."},{"key":"10.1016\/j.stem.2023.02.003_bib108","series-title":"Picard tools, Broad Institute, GitHub Repository","year":"2017"},{"key":"10.1016\/j.stem.2023.02.003_bib109","doi-asserted-by":"crossref","first-page":"81","DOI":"10.1007\/978-1-4939-0512-6_4","article-title":"Use model-based analysis of ChIP-Seq (MACS) to analyze short reads generated by sequencing protein-DNA interactions in embryonic stem cells","volume":"1150","author":"Liu","year":"2014","journal-title":"Methods Mol. Biol."},{"key":"10.1016\/j.stem.2023.02.003_bib110","series-title":"DiffBind: Differential Binding Analysis of ChIPSeq Peak Data","author":"Brown","year":"2022"},{"key":"10.1016\/j.stem.2023.02.003_bib111","doi-asserted-by":"crossref","first-page":"W160","DOI":"10.1093\/nar\/gkw257","article-title":"deepTools2: a next generation web server for deep-sequencing data analysis","volume":"44","author":"Ram\u00edrez","year":"2016","journal-title":"Nucleic Acids Res."},{"key":"10.1016\/j.stem.2023.02.003_bib112","doi-asserted-by":"crossref","first-page":"9354","DOI":"10.1038\/s41598-019-45839-z","article-title":"The ENCODE blacklist: identification of problematic regions of the genome","volume":"9","author":"Amemiya","year":"2019","journal-title":"Sci. Rep."},{"key":"10.1016\/j.stem.2023.02.003_bib113","doi-asserted-by":"crossref","first-page":"100052","DOI":"10.1016\/j.xpro.2020.100052","article-title":"Protocol for the generation of human pluripotent reporter cell lines using CRISPR\/Cas9","volume":"1","author":"Zhong","year":"2020","journal-title":"Star Protoc."},{"key":"10.1016\/j.stem.2023.02.003_bib114","doi-asserted-by":"crossref","first-page":"507","DOI":"10.1053\/j.gastro.2019.04.022","article-title":"Effects of serotonin and slow-release 5-hydroxytryptophan on gastrointestinal motility in a mouse model of depression","volume":"157","author":"Israelyan","year":"2019","journal-title":"Gastroenterology"},{"key":"10.1016\/j.stem.2023.02.003_bib115","doi-asserted-by":"crossref","first-page":"2221","DOI":"10.1172\/JCI84877","article-title":"Serotonin transporter variant drives preventable gastrointestinal abnormalities in development and function","volume":"126","author":"Margolis","year":"2016","journal-title":"J.\u00a0Clin. Invest."},{"key":"10.1016\/j.stem.2023.02.003_bib116","first-page":"53828","article-title":"Video imaging and spatiotemporal maps to analyze gastrointestinal motility in mice","volume":"108","author":"Swaminathan","year":"2016","journal-title":"J. Vis. Exp."},{"key":"10.1016\/j.stem.2023.02.003_bib117","doi-asserted-by":"crossref","first-page":"G1042","DOI":"10.1152\/ajpgi.00088.2020","article-title":"Antibiotic exposure postweaning disrupts the neurochemistry and function of enteric neurons mediating colonic motor activity","volume":"318","author":"Hung","year":"2020","journal-title":"Am. J. Physiol. Gastrointest. Liver Physiol."},{"key":"10.1016\/j.stem.2023.02.003_bib118","doi-asserted-by":"crossref","first-page":"167","DOI":"10.1002\/cyto.a.20022","article-title":"Design and validation of a tool for neurite tracing and analysis in fluorescence microscopy images","volume":"58","author":"Meijering","year":"2004","journal-title":"Cytometry A"}],"container-title":["Cell Stem Cell"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1934590923000401?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1934590923000401?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,30]],"date-time":"2025-10-30T13:56:38Z","timestamp":1761832598000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1934590923000401"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,3]]},"references-count":121,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2023,3]]}},"alternative-id":["S1934590923000401"],"URL":"https:\/\/doi.org\/10.1016\/j.stem.2023.02.003","relation":{"has-review":[{"id-type":"doi","id":"10.3410\/f.742557753.793598095","asserted-by":"object"}]},"ISSN":["1934-5909"],"issn-type":[{"value":"1934-5909","type":"print"}],"subject":[],"published":{"date-parts":[[2023,3]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"hPSC-derived sacral neural crest enables rescue in a severe model of Hirschsprung\u2019s disease","name":"articletitle","label":"Article Title"},{"value":"Cell Stem Cell","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.stem.2023.02.003","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2023 The Authors. Published by Elsevier Inc.","name":"copyright","label":"Copyright"}]}}