@article{9c83d5ace6e64fbe85fb555245390b77,
title = "Unbiased characterization of the larval zebrafish enteric nervous system at a single cell transcriptomic level",
abstract = "The enteric nervous system (ENS) regulates many gastrointestinal functions including peristalsis, immune regulation and uptake of nutrients. Defects in the ENS can lead to severe enteric neuropathies such as Hirschsprung disease (HSCR). Zebrafish have proven to be fruitful in the identification of genes involved in ENS development and HSCR pathogenesis. However, composition and specification of enteric neurons and glial subtypes at larval stages, remains mainly unexplored. Here, we performed single cell RNA sequencing of zebrafish ENS at 5 days post-fertilization. We identified vagal neural crest progenitors, Schwann cell precursors, and four clusters of differentiated neurons. In addition, a previously unrecognized elavl3+/phox2bb-population of neurons and cx43+/phox2bb-enteric glia was found. Pseudotime analysis supported binary neurogenic branching of ENS differentiation, driven by a notch-responsive state. Taken together, we provide new insights on ENS development and specification, proving that the zebrafish is a valuable model for the study of congenital enteric neuropathies.",
keywords = "Developmental neuroscience, Neuroscience, Transcriptomics",
author = "Kuil, {Laura E.} and Kakiailatu, {Naomi J.M.} and Windster, {Jonathan D.} and Eric Bindels and Zink, {Joke T.M.} and {van der Zee}, Gaby and Hofstra, {Robert M.W.} and Shepherd, {Iain T.} and Veerle Melotte and Alves, {Maria M.}",
note = "Funding Information: We want to thank Remco Hoogenboezem for mapping the scRNA-seq data, Emma de Pater for useful discussions, Soheil Yousefi for bioinformatics assistance, and the optical imaging center (OIC) of the Erasmus University Medical Center for assistance with confocal microscopy. Graphical abstract was created with BioRender.com. This work was funded by the Friends of the Sophia Foundation (SSWO WAR-63). R.M.W.H. M.M.A. and L.E.K. designed the study; L.E.K. N.J.M.K. I.T.S. E.B. J.T.M.Z. and G.Z. performed the experiments; L.E.K. and N.J.M.K. analyzed data, interpreted results, and prepared figures; L.E.K. and N.J.M.K. wrote the manuscript; M.M.A. and V.M. supervised the project; M.M.A. V.M. I.T.S. and J.D.W. revised the manuscript. All authors read and approved the final manuscript. The authors declare no competing interest, We support inclusive, diverse, and equitable conduct of research. During the preparation of this work ChatGPT - OpenAI is used to improve grammar. After using this tool, the authors reviewed and edited the content as needed and take full responsibility for the content of the publication. Funding Information: We want to thank Remco Hoogenboezem for mapping the scRNA-seq data, Emma de Pater for useful discussions, Soheil Yousefi for bioinformatics assistance, and the optical imaging center (OIC) of the Erasmus University Medical Center for assistance with confocal microscopy. Graphical abstract was created with BioRender.com. This work was funded by the Friends of the Sophia Foundation ( SSWO WAR-63 ). Publisher Copyright: {\textcopyright} 2023 The Author(s)",
year = "2023",
month = jul,
day = "21",
doi = "10.1016/j.isci.2023.107070",
language = "English",
volume = "26",
journal = "iScience",
issn = "2589-0042",
publisher = "Elsevier Inc.",
number = "7",
}