The transmembrane inner ear (tmie) gene contributes to vestibular and lateral line development and function in the zebrafish (Danio rerio)

Dev Dyn. 2008 Apr;237(4):941-52. doi: 10.1002/dvdy.21486.

Abstract

The inner ear is a complex organ containing sensory tissue, including hair cells, the development of which is not well understood. Our long-term goal is to discover genes critical for the correct formation and function of the inner ear and its sensory tissue. A novel gene, transmembrane inner ear (Tmie), was found to cause hearing-related disorders when defective in mice and humans. A homologous tmie gene in zebrafish was cloned and its expression characterized between 24 and 51 hours post-fertilization. Embryos injected with morpholinos (MO) directed against tmie exhibited circling swimming behavior (approximately 37%), phenocopying mice with Tmie mutations; semicircular canal formation was disrupted, hair cell numbers were reduced, and maturation of electrically active lateral line neuromasts was delayed. As in the mouse, tmie appears to be required for inner ear development and function in the zebrafish and for hair cell maturation in the vestibular and lateral line systems as well.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Behavior, Animal / physiology
  • Ear, Inner / anatomy & histology
  • Ear, Inner / embryology*
  • Ear, Inner / physiology*
  • Female
  • Gene Expression Regulation, Developmental
  • Humans
  • Lateral Line System / anatomy & histology
  • Lateral Line System / embryology*
  • Lateral Line System / physiology*
  • Male
  • Membrane Proteins / genetics*
  • Membrane Proteins / metabolism
  • Mice
  • Molecular Sequence Data
  • Morphogenesis
  • Oligonucleotides, Antisense / genetics
  • Oligonucleotides, Antisense / metabolism
  • Sequence Alignment
  • Swimming / physiology
  • Zebrafish Proteins / genetics*
  • Zebrafish Proteins / metabolism
  • Zebrafish* / anatomy & histology
  • Zebrafish* / embryology
  • Zebrafish* / physiology

Substances

  • Membrane Proteins
  • Oligonucleotides, Antisense
  • Tmie protein, zebrafish
  • Zebrafish Proteins