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

Journal Article

TítuloPhox2b function in the enteric nervous system is conserved in zebrafish and is sox10-dependent.
Publication TypeJournal Article
AuthorsElworthy, S, Pinto, JP, Pettifer, A, M. Cancela, L, Kelsh, RN
Year of Publication2005
JournalMech Dev
Date Published2005 May
Palavras-chaveAmino Acid Sequence, Animals, Carrier Proteins, Central Nervous System, Chromosome Mapping, Enteric Nervous System, Gene Expression Regulation, Developmental, High Mobility Group Proteins, Homeodomain Proteins, In Situ Hybridization, Melanophores, Microscopy, Fluorescence, Models, Genetic, Molecular Sequence Data, Mutation, Neural Crest, Neurons, Phenotype, Phylogeny, Sequence Homology, Amino Acid, SOXE Transcription Factors, Stem Cells, Time Factors, Transcription Factors, Zebrafish, Zebrafish Proteins

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.


Alternate JournalMech. Dev.
PubMed ID15817223
Grant ListG0300415 / / Medical Research Council / United Kingdom
CCMAR Authors