PUBLICATION
Small fish, big prospects: using zebrafish to unravel the mechanisms of hereditary hearing loss
- Authors
- Vona, B., Doll, J., Hofrichter, M.A.H., Haaf, T., Varshney, G.K.
- ID
- ZDB-PUB-200221-15
- Date
- 2020
- Source
- Hearing Research 397: 107906 (Review)
- Registered Authors
- Varshney, Gaurav
- Keywords
- Base editing, CRISPR/Cas9, Genetic variants, Genetics of hearing loss, Hereditary hearing loss, Model organism, Zebrafish
- MeSH Terms
-
- Animals
- CRISPR-Cas Systems
- Gene Editing
- Hearing/genetics
- Hearing Loss*/genetics
- Humans
- Zebrafish/genetics
- PubMed
- 32063424 Full text @ Hear. Res.
Citation
Vona, B., Doll, J., Hofrichter, M.A.H., Haaf, T., Varshney, G.K. (2020) Small fish, big prospects: using zebrafish to unravel the mechanisms of hereditary hearing loss. Hearing Research. 397:107906.
Abstract
Over the past decade, advancements in high-throughput sequencing have greatly enhanced our knowledge of the mutational signatures responsible for hereditary hearing loss. In its present state, the field has a largely uncensored view of protein coding changes in a growing number of genes that have been associated with hereditary hearing loss, and many more that have been proposed as candidate genes. Sequencing data can now be generated using methods that have become widespread and affordable. The greatest hurdles facing the field concern functional validation of uncharacterized genes and rapid application to human diseases, including hearing and balance disorders. To date, over 30 hearing-related disease models exist in zebrafish. New genome editing technologies, including CRISPR/Cas9 will accelerate the functional validation of hearing loss genes and variants in zebrafish. Here, we discuss current progress in the field and recent advances in genome editing approaches.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping