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Keywords: Hirschsprung disease
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Journal Articles

Endothelin signaling in development

Stanley M. Kanai, David E. Clouthier
Journal: Development
Development (2023) 150 (24): dev201786.
https://doi.org/10.1242/dev.201786
Published: 11 December 2023
... of endothelin signaling during craniofacial, cardiovascular, melanocyte and enteric nervous system development and describes the evolutionary conservation of endothelin action and implications to human health. G protein Craniofacial Cardiovascular Enteric Melanocyte Hirschsprung disease...
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Journal Articles

CXCR4 and CXCL12 signaling regulates the development of extrinsic innervation to the colorectum

Viktória Halasy, Emőke Szőcs, Ádám Soós, Tamás Kovács, Nóra Pecsenye-Fejszák, Ryo Hotta, Allan M. Goldstein, Nándor Nagy
Journal: Development
Development (2023) 150 (8): dev201289.
https://doi.org/10.1242/dev.201289
Published: 20 April 2023
... in formation of extrinsic innervation of the colorectum, the control of which is poorly understood. Hindgut Extrinsic innervation CXCR4 Enteric nervous system Neural crest cells Hirschsprung disease National Institutes of Health R01DK119210 R21HD106036 Innovációs és...
View article titled, CXCR4 and CXCL12 signaling regulates the development of extrinsic innervation to the colorectum
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Includes: Supplementary data
Journal Articles

A distinct transcriptome characterizes neural crest-derived cells at the migratory wavefront during enteric nervous system development

Rhian Stavely, Ryo Hotta, Richard A. Guyer, Nicole Picard, Ahmed A. Rahman, Meredith Omer, Adam Soos, Emoke Szocs, Jessica Mueller, Allan M. Goldstein, Nandor Nagy
Journal: Development
Development (2023) 150 (5): dev201090.
https://doi.org/10.1242/dev.201090
Published: 6 March 2023
... , H. M. ( 2009 ). The migratory behavior of immature enteric neurons . Dev. Neurobiol. 69 , 22 - 35 . 10.1002/dneu.20683 Heanue , T. A. and Pachnis , V. ( 2007 ). Enteric nervous system development and Hirschsprung's disease: advances in genetic and stem cell studies...
View article titled, A distinct transcriptome characterizes neural crest-derived cells at the migratory wavefront during enteric nervous system development
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Includes: Supplementary data
Journal Articles

Avian ceca are indispensable for hindgut enteric nervous system development

Nandor Nagy, Tamas Kovacs, Rhian Stavely, Viktoria Halasy, Adam Soos, Emoke Szocs, Ryo Hotta, Hannah Graham, Allan M. Goldstein
Journal: Development
Development (2021) 148 (22): dev199825.
https://doi.org/10.1242/dev.199825
Published: 26 November 2021
... migration can lead to Hirschsprung disease, which is characterized by an aganglionic distal bowel. During the craniocaudal migration of ENCCs along the gut, we find that their proliferation is greatest as the ENCC wavefront passes through the ceca, a pair of pouches at the midgut-hindgut junction in avian...
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Includes: Supplementary data
Journal Articles

RET overactivation leads to concurrent Hirschsprung disease and intestinal ganglioneuromas

Nandor Nagy, Richard A. Guyer, Ryo Hotta, Dongcheng Zhang, Donald F. Newgreen, Viktoria Halasy, Tamas Kovacs, Allan M. Goldstein
Journal: Development
Development (2020) 147 (21): dev190900.
https://doi.org/10.1242/dev.190900
Published: 5 November 2020
... and differentiation. RET deficiency, for example, leads to intestinal aganglionosis (Hirschsprung disease), whereas overactive RET can lead to multiple endocrine neoplasia (MEN) syndromes. Some RET mutations are associated with both intestinal aganglionosis and MEN-associated tumors. This seemingly paradoxical...
View article titled, RET overactivation leads to concurrent <span class="search-highlight">Hirschsprung</span> <span class="search-highlight">disease</span> and intestinal ganglioneuromas
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Includes: Supplementary data
Journal Articles

Collagen 18 and agrin are secreted by neural crest cells to remodel their microenvironment and regulate their migration during enteric nervous system development

Nandor Nagy, Csilla Barad, Ryo Hotta, Sukhada Bhave, Emily Arciero, David Dora, Allan M. Goldstein
Journal: Development
Development (2018) 145 (9): dev160317.
https://doi.org/10.1242/dev.160317
Published: 8 May 2018
...-dependent pathogenic mechanism for Hirschsprung's disease . J. Clin. Invest.   125 , 4483 - 4496 . 10.1172/JCI83178 Su , J. , Stenbjorn , R. S. , Gorse , K. , Su , K. , Hauser , K. F. , Ricard-Blum , S. , Pihlajaniemi , T. and Fox , M. A. ( 2012 ). Target-derived...
View article titled, Collagen 18 and agrin are secreted by neural crest cells to remodel their microenvironment and regulate their migration during enteric nervous system development
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Includes: Supplementary data
Journal Articles

Sonic hedgehog controls enteric nervous system development by patterning the extracellular matrix

Nandor Nagy, Csilla Barad, Hannah K. Graham, Ryo Hotta, Lily S. Cheng, Nora Fejszak, Allan M. Goldstein
Journal: Development
Development (2016) 143 (2): 264–275.
https://doi.org/10.1242/dev.128132
Published: 15 January 2016
... enteric nervous system by regulating the composition of the intestinal microenvironment. Enteric nervous system Sonic hedgehog Patched Ptch Epithelium Extracellular matrix Enteric neural crest cells Hirschsprung disease Chick Quail Mouse Gut development relies on interactions among...
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Includes: Supplementary data
Journal Articles

Vitamin A facilitates enteric nervous system precursor migration by reducing Pten accumulation

Ming Fu, Yoshiharu Sato, Ariel Lyons-Warren, Bin Zhang, Maureen A. Kane, Joseph L. Napoli, Robert O. Heuckeroth
Journal: Development
Development (2010) 137 (4): 631–640.
https://doi.org/10.1242/dev.040550
Published: 15 February 2010
...Ming Fu; Yoshiharu Sato; Ariel Lyons-Warren; Bin Zhang; Maureen A. Kane; Joseph L. Napoli; Robert O. Heuckeroth Hirschsprung disease is a serious disorder of enteric nervous system (ENS) development caused by the failure of ENS precursor migration into the distal bowel. We now demonstrate...
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Includes: Supplementary data
Journal Articles

Mice expressing a dominant-negative Ret mutation phenocopy human Hirschsprung disease and delineate a direct role of Ret in spermatogenesis

Sanjay Jain, Cathy K. Naughton, Mao Yang, Amy Strickland, Kiran Vij, Mario Encinas, Judy Golden, Akshay Gupta, Robert Heuckeroth, Eugene M. Johnson, Jr, Jeffrey Milbrandt
Journal: Development
Development (2004) 131 (21): 5503–5513.
https://doi.org/10.1242/dev.01421
Published: 1 November 2004
... activation of AKT. All Ret DN/+ mice died by 1 month of age and had distal intestinal aganglionosis reminiscent of Hirschsprung disease (HSCR) in humans. The Ret DN/+ proximal small intestine also had severe hypoganglionosis and reduction in nerve fiber density, suggesting a potential mechanism...
View article titled, Mice expressing a dominant-negative Ret mutation phenocopy human <span class="search-highlight">Hirschsprung</span> <span class="search-highlight">disease</span> and delineate a direct role of Ret in spermatogenesis
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Includes: Supplementary data
Journal Articles

A transgenic model for studying development of the enteric nervous system in normal and aganglionic mice

Raj P. Kapur, Cynthia Yost, Richard D. Palmiter
Journal: Development
Development (1992) 116 (1): 167–175.
https://doi.org/10.1242/dev.116.1.167
Published: 1 September 1992
... of non-catecholaminergic enteric neurons . Devl. Biol . 141 , 353 – 380 . Bolande , R. P. ( 1975 ). Animal model of human disease. Hirschsprung’s disease, aganglionic or hypoganglionic megacolon; animal model: aganglionic megacolon in piebald and spotted mutant mouse strains . Am. J...
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