Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent

Stone Elworthy, Jorge P. Pinto, Anna Pettifer, M. Leonor Cancela, Robert N. Kelsh

Research output: Contribution to journalArticle

  • 68 Citations

Abstract

Zebrafish lacking functional sox10 have defects in non-ectomesenchymal neural crest derivatives including the enteric nervous system (ENS) and as such provide an animal model for human Waardenburg Syndrome IV. Here, we characterize zebrafish phox2b as a functionally conserved marker of the developing ENS. We show that morpholino-mediated knockdown of Phox2b generates fish modeling Hirschsprung disease. Using markers, including phox2b, we investigate the ontogeny of the sox10 ENS phenotype. As previously shown for melanophore development, ENS progenitor fate specification fails in these mutant fish. However, in addition, we trace back the sox10 mutant ENS defect to an even earlier time point, finding that most neural crest cells fail to migrate ventrally to the gut primordium. (c) 2005 Elsevier Ireland Ltd. All rights reserved.
LanguageEnglish
Pages659-669
JournalMechanisms of Development
Volume122
Issue number5
DOIs
StatusPublished - 2005

Fingerprint

Enteric Nervous System
Zebrafish
Neural Crest
Fishes
Waardenburg Syndrome
Melanophores
Morpholinos
Hirschsprung Disease
Ireland
Animal Models
Phenotype

Cite this

Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent. / Elworthy, Stone; Pinto, Jorge P.; Pettifer, Anna; Cancela, M. Leonor; Kelsh, Robert N.

In: Mechanisms of Development, Vol. 122, No. 5, 2005, p. 659-669.

Research output: Contribution to journalArticle

Elworthy, Stone ; Pinto, Jorge P. ; Pettifer, Anna ; Cancela, M. Leonor ; Kelsh, Robert N./ Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent. In: Mechanisms of Development. 2005 ; Vol. 122, No. 5. pp. 659-669
@article{0fe2cf592f664e299c231f1e81f0e82d,
title = "Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent",
abstract = "Zebrafish lacking functional sox10 have defects in non-ectomesenchymal neural crest derivatives including the enteric nervous system (ENS) and as such provide an animal model for human Waardenburg Syndrome IV. Here, we characterize zebrafish phox2b as a functionally conserved marker of the developing ENS. We show that morpholino-mediated knockdown of Phox2b generates fish modeling Hirschsprung disease. Using markers, including phox2b, we investigate the ontogeny of the sox10 ENS phenotype. As previously shown for melanophore development, ENS progenitor fate specification fails in these mutant fish. However, in addition, we trace back the sox10 mutant ENS defect to an even earlier time point, finding that most neural crest cells fail to migrate ventrally to the gut primordium. (c) 2005 Elsevier Ireland Ltd. All rights reserved.",
author = "Stone Elworthy and Pinto, {Jorge P.} and Anna Pettifer and Cancela, {M. Leonor} and Kelsh, {Robert N.}",
year = "2005",
doi = "10.1016/j.mod.2004.12.008",
language = "English",
volume = "122",
pages = "659--669",
journal = "Mechanisms of Development",
issn = "0925-4773",
publisher = "Elsevier Ireland Ltd",
number = "5",

}

TY - JOUR

T1 - Phox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent

AU - Elworthy,Stone

AU - Pinto,Jorge P.

AU - Pettifer,Anna

AU - Cancela,M. Leonor

AU - Kelsh,Robert N.

PY - 2005

Y1 - 2005

N2 - Zebrafish lacking functional sox10 have defects in non-ectomesenchymal neural crest derivatives including the enteric nervous system (ENS) and as such provide an animal model for human Waardenburg Syndrome IV. Here, we characterize zebrafish phox2b as a functionally conserved marker of the developing ENS. We show that morpholino-mediated knockdown of Phox2b generates fish modeling Hirschsprung disease. Using markers, including phox2b, we investigate the ontogeny of the sox10 ENS phenotype. As previously shown for melanophore development, ENS progenitor fate specification fails in these mutant fish. However, in addition, we trace back the sox10 mutant ENS defect to an even earlier time point, finding that most neural crest cells fail to migrate ventrally to the gut primordium. (c) 2005 Elsevier Ireland Ltd. All rights reserved.

AB - Zebrafish lacking functional sox10 have defects in non-ectomesenchymal neural crest derivatives including the enteric nervous system (ENS) and as such provide an animal model for human Waardenburg Syndrome IV. Here, we characterize zebrafish phox2b as a functionally conserved marker of the developing ENS. We show that morpholino-mediated knockdown of Phox2b generates fish modeling Hirschsprung disease. Using markers, including phox2b, we investigate the ontogeny of the sox10 ENS phenotype. As previously shown for melanophore development, ENS progenitor fate specification fails in these mutant fish. However, in addition, we trace back the sox10 mutant ENS defect to an even earlier time point, finding that most neural crest cells fail to migrate ventrally to the gut primordium. (c) 2005 Elsevier Ireland Ltd. All rights reserved.

UR - http://dx.doi.org/10.1016/j.mod.2004.12.008

U2 - 10.1016/j.mod.2004.12.008

DO - 10.1016/j.mod.2004.12.008

M3 - Article

VL - 122

SP - 659

EP - 669

JO - Mechanisms of Development

T2 - Mechanisms of Development

JF - Mechanisms of Development

SN - 0925-4773

IS - 5

ER -