Distinct Functional and Temporal Requirements for Zebrafish Hdac1 during Neural Crest-Derived Craniofacial and Peripheral Neuron Development

Ignatius, M.S., Unal Eroglu, A., Malireddy, S., Gallagher, G., Nambiar, R.M., and Henion, P.D.
PLoS One   8(5): e63218 (Journal)
Registered Authors
Gallagher, Glen, Henion, Paul, Ignatius, Myron, Malireddy, Smitha, Nambiar, Roopa
MeSH Terms
  • Animals
  • Branchial Region/abnormalities
  • Branchial Region/embryology
  • Branchial Region/pathology
  • Cell Differentiation/drug effects
  • Craniofacial Abnormalities/embryology
  • Craniofacial Abnormalities/pathology
  • Embryo, Nonmammalian/drug effects
  • Embryo, Nonmammalian/metabolism
  • Embryo, Nonmammalian/pathology
  • Face/abnormalities
  • Face/embryology*
  • Face/pathology
  • Histone Deacetylase 1/genetics
  • Histone Deacetylase 1/metabolism*
  • Hydroxamic Acids/pharmacology
  • Hyoid Bone/abnormalities
  • Hyoid Bone/drug effects
  • Hyoid Bone/embryology
  • Hyoid Bone/pathology
  • Mandible/abnormalities
  • Mandible/drug effects
  • Mandible/embryology
  • Mandible/pathology
  • Mutation/genetics
  • Neural Crest/drug effects
  • Neural Crest/embryology
  • Neural Crest/metabolism
  • Neural Crest/pathology*
  • Neurons/drug effects
  • Neurons/metabolism*
  • Neurons/pathology
  • Peripheral Nervous System/drug effects
  • Peripheral Nervous System/embryology
  • Peripheral Nervous System/pathology
  • Phenotype
  • Skull/abnormalities
  • Skull/embryology*
  • Skull/pathology
  • Stem Cells/drug effects
  • Stem Cells/metabolism
  • Stem Cells/pathology
  • Sympathetic Nervous System/drug effects
  • Sympathetic Nervous System/metabolism
  • Sympathetic Nervous System/pathology
  • Time Factors
  • Zebrafish/embryology*
  • Zebrafish/metabolism*
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism*
23667588 Full text @ PLoS One

The regulation of gene expression is accomplished by both genetic and epigenetic means and is required for the precise control of the development of the neural crest. In hdac1b382 mutants, craniofacial cartilage development is defective in two distinct ways. First, fewer hoxb3a, dlx2 and dlx3-expressing posterior branchial arch precursors are specified and many of those that are consequently undergo apoptosis. Second, in contrast, normal numbers of progenitors are present in the anterior mandibular and hyoid arches, but chondrocyte precursors fail to terminally differentiate. In the peripheral nervous system, there is a disruption of enteric, DRG and sympathetic neuron differentiation in hdac1b382 mutants compared to wildtype embryos. Specifically, enteric and DRG-precursors differentiate into neurons in the anterior gut and trunk respectively, while enteric and DRG neurons are rarely present in the posterior gut and tail. Sympathetic neuron precursors are specified in hdac1b382 mutants and they undergo generic neuronal differentiation but fail to undergo noradrenergic differentiation. Using the HDAC inhibitor TSA, we isolated enzyme activity and temporal requirements for HDAC function that reproduce hdac1b382 defects in craniofacial and sympathetic neuron development. Our study reveals distinct functional and temporal requirements for zebrafish hdac1 during neural crest-derived craniofacial and peripheral neuron development.

Genes / Markers
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Engineered Foreign Genes
Errata and Notes