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Ankfn1 -mutant vestibular defects require loss of both ancestral and derived paralogs for penetrance in zebrafish
Open accessPublished version of a preprint
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.
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Metadata
- Authors
- Ross, K. D., Ren, J., Zhang, R., Chi, N. C., & Hamilton, B. A.
- Affiliations
- University of California San Diego (US)
- Biomedical Sciences Graduate Program, University of California San Diego, La Jolla, CA 92093, USA
- Corresponding author
- Hamilton, Bruce A
- Venue
- G3 Genes|Genomes|Genetics
- Publisher
- Oxford University Press (OUP)
- Published
- 2021-12-25
- Volume
- 12
- Issue
- 3
- ISSN
- 2160-1836 (ISSN-L)
- PMC
- PMC9210315
- OpenAlex
- W4200434406
- Cited by
- 4
- References
- 2
- Citations by year
- 2026: 2 · 2025: 1 · 2024: 1
- FWCI
- 0.08
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- G3 Genes Genomes Genetics (publishedVersion, cc-by)
- bioRxiv (Cold Spring Harbor Laboratory) (acceptedVersion, cc-by-nd)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- Funders
- National Institutes of Health (R01 GM086912 and R01 NS097534 to BAH)
- Ruth L. Kirschstein Institutional National Research Service (T32 GM008666)
- National Institute for General Medical Sciences
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Life Sciences › Neuroscience › Neurology › Vestibular and auditory disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Zebrafish Biomedical Research Applications
- Keywords
- gene duplication, gene dosage, zebrafish, evolutionary innovation, neofunctionalization, sensorimotor development
- MeSH terms
- Animals, Phylogeny, Zebrafish, Evolution, Molecular, Penetrance, Gene Duplication, Organogenesis, Mice
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