PUBLICATION

Zebrafish models of human eye and inner ear diseases

Authors
Blanco-Sánchez, B., Clément, A., Phillips, J.B., Westerfield, M.
ID
ZDB-PUB-170129-13
Date
2017
Source
Methods in cell biology   138: 415-467 (Chapter)
Registered Authors
Blanco, Bernardo, Clément, Aurélie, Phillips, Jennifer, Westerfield, Monte
Keywords
Deafness, Hair cell, Human disease, Mechanosensory cell, Photoreceptor, Retina, Retinal degeneration, Retinitis pigmentosa, Usher syndrome
MeSH Terms
  • Animals
  • Disease Models, Animal
  • Eye/growth & development*
  • Eye/physiopathology
  • Gene Editing/methods*
  • Humans
  • Labyrinth Diseases/genetics*
  • Labyrinth Diseases/physiopathology
  • Mutation
  • Phenotype
  • Zebrafish/genetics*
  • Zebrafish/growth & development
PubMed
28129854 Full text @ Meth. Cell. Biol.
Abstract
Eye and inner ear diseases are the most common sensory impairments that greatly impact quality of life. Zebrafish have been intensively employed to understand the fundamental mechanisms underlying eye and inner ear development. The zebrafish visual and vestibulo-acoustic systems are very similar to these in humans, and although not yet mature, they are functional by 5days post-fertilization (dpf). In this chapter, we show how the zebrafish has significantly contributed to the field of biomedical research and how researchers, by establishing disease models and meticulously characterizing their phenotypes, have taken the first steps toward therapies. We review here models for (1) eye diseases, (2) ear diseases, and (3) syndromes affecting eye and/or ear. The use of new genome editing technologies and high-throughput screening systems should increase considerably the speed at which knowledge from zebrafish disease models is acquired, opening avenues for better diagnostics, treatments, and therapies.
Genes / Markers
Figures
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping