PUBLICATION
Ankfn1-mutant vestibular defects require loss of both ancestral and derived paralogs for penetrance in zebrafish
- Authors
- Ross, K.D., Ren, J., Zhang, R., Chi, N.C., Hamilton, B.A.
- ID
- ZDB-PUB-220202-29
- Date
- 2021
- Source
- G3 (Bethesda) 12(3): (Journal)
- Registered Authors
- Chi, Neil C.
- Keywords
- Ankfn1, Ankfn1-like, Banderuola, nmf9, wide awake, domain architecture, paralogy, penetrance
- MeSH Terms
-
- Animals
- Evolution, Molecular*
- Gene Duplication
- Mice
- Organogenesis
- Penetrance
- Phylogeny
- Zebrafish*/genetics
- PubMed
- 35100349 Full text @ G3 (Bethesda)
Citation
Ross, K.D., Ren, J., Zhang, R., Chi, N.C., Hamilton, B.A. (2021) Ankfn1-mutant vestibular defects require loss of both ancestral and derived paralogs for penetrance in zebrafish. G3 (Bethesda). 12(3):.
Abstract
How and to what degree gene duplication events create regulatory innovation, redundancy, or neofunctionalization remain important questions in animal evolution and comparative genetics. Ankfn1 genes are single copy in most invertebrates, partially duplicated in jawed vertebrates, and only the derived copy retained in most mammals. Null mutations in the single mouse homolog have vestibular and neurological abnormalities. Null mutation of the single Drosophila homolog is typically lethal with severe sensorimotor deficits in rare survivors. The functions and potential redundancy of paralogs in species with two copies are not known. Here, we define a vestibular role for Ankfn1 homologs in zebrafish based on the simultaneous disruption of each locus. Zebrafish with both paralogs disrupted showed vestibular defects and early lethality from swim bladder inflation failure. One intact copy at either locus was sufficient to prevent major phenotypes. Our results show that vertebrate Ankfn1 genes are required for vestibular-related functions, with at least partial redundancy between ancestral and derived paralogs.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping