title: Zebrafish models of human eye and inner ear diseases.
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.
authors.affiliation: University of Oregon, Eugene, OR, United States.
authors.forename: B
authors.initials: B
authors.lastname: Blanco-Sánchez
authors.affiliation: University of Oregon, Eugene, OR, United States.
authors.forename: A
authors.initials: A
authors.lastname: Clément
authors.affiliation: University of Oregon, Eugene, OR, United States.
authors.forename: J B
authors.initials: JB
authors.lastname: Phillips
authors.affiliation: University of Oregon, Eugene, OR, United States.
authors.forename: M
authors.initials: M
authors.lastname: Westerfield
mesh_terms: D000818:Animals; D004195:Disease Models, Animal; D005123:Eye / Q000254:growth & development* / Q000503:physiopathology; D000072669:Gene Editing / Q000379:methods*; D006801:Humans; D007759:Labyrinth Diseases / Q000235:genetics* / Q000503:physiopathology; D009154:Mutation; D010641:Phenotype; D015027:Zebrafish / Q000235:genetics* / Q000254:growth & development
keywords: Deafness; Hair cell; Human disease; Mechanosensory cell; Photoreceptor; Retina; Retinal degeneration; Retinitis pigmentosa; Usher syndrome