ArticleMolecular and cellular biology2015
Otoferlin deficiency in zebrafish results in defects in balance and hearing: rescue of the balance and hearing phenotype with full-length and truncated forms of mouse otoferlin.
Article in Molecular and cellular biology, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers.
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Who cites it
32 citing papers in PubMed, 51 citations in OpenAlex.
- Electrophysiological Profiling of Exocytosis during Early-Stage Development of the Zebrafish Lateral Line.eNeuro · 2026Article
- Zebrafish pou3f3b controls saccular/auditory development and marks non-neuronal cells that delaminate from the otic vesicle to promote neuroblast maturation.Developmental biology · 2026Article
- Electrophysiological profiling of exocytosis during early-stage development of the zebrafish lateral line.bioRxiv : the preprint server for biology · 2025Article
- Expression of nestin, parvalbumin and otoferlin during cochlear development in the mouse: an immunofluorescence study.European journal of histochemistry : EJH · 2025Article
- Cochlear gene therapy restores hearing and auditory processing in an atypical DFNB9 mouse model.Communications medicine · 2025Article
- Article
- Probing the role of the CFrontiers in molecular neuroscience · 2023Article
- Otoferlin as a multirole CaFrontiers in cellular neuroscience · 2023Review
- shox2 is required for vestibular statoacoustic neuron development.Biology open · 2022Article
- Chronic neurotransmission increases the susceptibility of lateral-line hair cells to ototoxic insults.eLife · 2022Article
- Co-transduction of dual-adeno-associated virus vectors in the neonatal and adult mouse utricles.Frontiers in molecular neuroscience · 2022Article
- Truncation of the otoferlin transmembrane domain alters the development of hair cells and reduces membrane docking.Molecular biology of the cell · 2021Article
- How Zebrafish Can Drive the Future of Genetic-based Hearing and Balance Research.Journal of the Association for Research in Otolaryngology : JARO · 2021Review
- Whole-Exome Sequencing Reveals a Rare Variant ofFrontiers in genetics · 2021Article
- Small fish, big prospects: using zebrafish to unravel the mechanisms of hereditary hearing loss.Hearing research · 2020Article
- Review
- Transcriptomic Analyses of Inner Ear Sensory Epithelia in Zebrafish.Anatomical record (Hoboken, N.J. : 2007) · 2020Article
- Functions of Vertebrate Ferlins.Cells · 2020Review
- Otoferlin C2F Domain-Induced Changes in Membrane Structure Observed by Sum Frequency Generation.Biophysical journal · 2019Article
- Otoferlin Depletion Results in Abnormal Synaptic Ribbons and Altered Intracellular Calcium Levels in Zebrafish.Scientific reports · 2019Article
Corrections and comments
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
Sensory hair cells convert mechanical motion into chemical signals. Otoferlin, a six-C2 domain transmembrane protein linked to deafness in humans, is hypothesized to play a role in exocytosis at hair cell ribbon synapses. To date, however, otoferlin has been studied almost exclusively in mouse models, and no rescue experiments have been reported. Here we describe the phenotype associated with morpholino-induced otoferlin knockdown in zebrafish and report the results of rescue experiments conducted with full-length and truncated forms of otoferlin. We found that expression of otoferlin occurs early in development and is restricted to hair cells and the midbrain. Immunofluorescence microscopy revealed localization to both apical and basolateral regions of hair cells. Knockdown of otoferlin resulted in hearing and balance defects, as well as locomotion deficiencies. Further, otoferlin morphants had uninflated swim bladders. Rescue experiments conducted with mouse otoferlin restored hearing, balance, and inflation of the swim bladder. Remarkably, truncated forms of otoferlin retaining the C-terminal C2F domain also rescued the otoferlin knockdown phenotype, while the individual N-terminal C2A domain did not. We conclude that otoferlin plays an evolutionarily conserved role in vertebrate hearing and that truncated forms of otoferlin can rescue hearing and balance.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.