PUBLICATION

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

Authors

Elworthy, S., Pinto, J.P., Pettifer, A., Cancela, M.L., and Kelsh, R.N.

ID

ZDB-PUB-050413-8

Date

2005

Source

Mechanisms of Development 122(5): 659-669 (Journal)

Registered Authors

Elworthy, Stone, Kelsh, Robert

Keywords

Phox2b; Enteric neuron; Colorless; Sox10; Fate specification; Progenitors; Stem cells; Neural crest; Melanophore

MeSH Terms

Amino Acid Sequence
Models, Genetic
Chromosome Mapping
High Mobility Group Proteins/genetics*
High Mobility Group Proteins/metabolism
High Mobility Group Proteins/physiology*
Zebrafish
Homeodomain Proteins/metabolism
Homeodomain Proteins/physiology*
Melanophores/metabolism
Molecular Sequence Data
Transcription Factors/metabolism
Transcription Factors/physiology*
SOXE Transcription Factors
Gene Expression Regulation, Developmental*
Neural Crest/metabolism
Sequence Homology, Amino Acid
Central Nervous System/embryology
Zebrafish Proteins/genetics*
Zebrafish Proteins/metabolism
Zebrafish Proteins/physiology*
Carrier Proteins/genetics*
Carrier Proteins/metabolism
Carrier Proteins/physiology*
Animals
Neurons/metabolism
Phylogeny
Time Factors
Phenotype
In Situ Hybridization
Mutation
Stem Cells/metabolism
Microscopy, Fluorescence
Enteric Nervous System/embryology*

(all 34)

PubMed

15817223 Full text @ Mech. Dev.

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.

Genes / Markers

Figures

Show all Figures

Expression

Gene	Fish	Conditions	Stage	Qualifier	Anatomy	Assay	Figure
crestin	sox10^m618/m618 (AB)	standard conditions	Prim-5		neural crest	ISH	Fig. 8
crestin	WT	standard conditions	Prim-5		neural crest	ISH	Fig. 8
phox2bb	sox10^m618/m618 (AB)	control	Pec-fin	Not Detected	enteric nervous system	ISH	Fig. 7
phox2bb	sox10^m618/m618 (AB)	standard conditions	Pec-fin		epibranchial placode	ISH	Fig. 2
phox2bb	sox10^m618/m618 (AB)	control	Prim-15		hindbrain	ISH	Fig. 7
phox2bb	sox10^m618/m618 (AB)	control	High-pec		mesenchyme pectoral fin	ISH	Fig. 7
phox2bb	sox10^m618/m618 (AB)	control	Prim-15	Not Detected	presumptive enteric nervous system	ISH	Fig. 7
phox2bb	sox10^m618/m618 (AB)	control	High-pec	Not Detected	presumptive enteric nervous system	ISH	Fig. 7
phox2bb	WT	control	Pec-fin		enteric nervous system	ISH	Fig. 7
phox2bb	WT	standard conditions	Pec-fin		enteric nervous system	ISH	Fig. 3

1 - 10 of 38

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Phenotype

Fish	Conditions	Stage	Phenotype	Figure
WT + MO1-phox2bb	standard conditions	Day 5	enteric nervous system neuron decreased amount, abnormal	Fig. 5
WT + MO2-phox2bb	standard conditions	Day 5	enteric nervous system neuron decreased amount, abnormal	text only
sox10^m618/m618 (AB)	control	Prim-15	presumptive enteric nervous system aplastic, abnormal	Fig. 7
sox10^m618/m618 (AB)	control	High-pec	presumptive enteric nervous system aplastic, abnormal	Fig. 7
sox10^m618/m618 (AB)	control	Pec-fin	presumptive enteric nervous system aplastic, abnormal	Fig. 7

1 - 5 of 5

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
m618		Point Mutation	sox10
tw11		Point Mutation	sox10

Human Disease / Model

Sequence Targeting Reagents

Target	Reagent	Reagent Type
phox2bb	MO1-phox2bb	MRPHLNO
phox2bb	MO2-phox2bb	MRPHLNO

Fish

Fish
sox10^m618/m618 (AB)
sox10^tw11/tw11
WT
WT + MO1-phox2bb
WT + MO2-phox2bb

Antibodies

Orthology

Gene	Orthology
phox2bb

Engineered Foreign Genes

Mapping

Entity Type	Entity Symbol	Location
GENE	phox2bb	Chr: 14 Details
SSLP	z6847	Chr: 14 Details

Elworthy et al., 2005 ZDB-PUB-050413-8 PMID:15817223

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

Elworthy et al., 2005

ZDB-PUB-050413-8
PMID:15817223