PUBLICATION

Vitamin D receptor deficiency impairs inner ear development in zebrafish

Authors
Kwon, H.J.
ID
ZDB-PUB-160818-14
Date
2016
Source
Biochemical and Biophysical Research Communications   478(2): 994-8 (Journal)
Registered Authors
Kwon, Hye-Joo
Keywords
Ear development, Vitamin D, Vitamin D receptor (VDR), Zebrafish
MeSH Terms
  • Animals
  • Cell Count
  • Ear, Inner/embryology*
  • Ear, Inner/metabolism*
  • Ear, Inner/pathology
  • Embryo, Nonmammalian/metabolism
  • Embryo, Nonmammalian/pathology
  • Embryonic Development
  • Gene Knockdown Techniques
  • Hair Cells, Auditory/pathology
  • Phenotype
  • Receptors, Calcitriol/deficiency*
  • Receptors, Calcitriol/metabolism*
  • Zebrafish/embryology*
  • Zebrafish/metabolism*
PubMed
27526995 Full text @ Biochem. Biophys. Res. Commun.
Abstract
The biological actions of vitamin D are largely mediated through binding to the vitamin D receptor (VDR), a member of the nuclear hormone receptor family, which regulates gene expression in a wide variety of tissues and cells. Mutations in VDR gene have been implicated in ear disorders (hearing loss and balance disorder) but the mechanisms are not well established. In this study, to investigate the role of VDR in inner ear development, morpholino-mediated gene knockdown approaches were used in zebrafish model system. Two paralogs for VDR, vdra and vdrb, have been identified in zebrafish. Knockdown of vdra had no effect on ear development, whereas knockdown of vdrb displayed morphological ear defects including smaller otic vesicles with malformed semicircular canals and abnormal otoliths. Loss-of-vdrb resulted in down-regulation of pre-otic markers, pax8 and pax2a, indicating impairment of otic induction. Furthermore, zebrafish embryos lacking vdrb produced fewer sensory hair cells in the ears and showed disruption of balance and motor coordination. These data reveal that VDR signaling plays an important role in ear development.
Genes / Markers
Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes