ZFIN ID: ZDB-PUB-160818-14
Vitamin D receptor deficiency impairs inner ear development in zebrafish
Kwon, H.J.
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.
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.