ArticleDevelopment (Cambridge, England)2009
Nkcc1 (Slc12a2) is required for the regulation of endolymph volume in the otic vesicle and swim bladder volume in the zebrafish larva.
Article in Development (Cambridge, England), 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 36 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
36 citing papers in PubMed, 59 citations in OpenAlex.
- Hydrostatic pressure shapes and canalizes semicircular canal morphology to ensure vestibular function.bioRxiv : the preprint server for biology · 2026Article
- Development of the semicircular canals and otolithic organs of the vertebrate inner ear.Current topics in developmental biology · 2025Review
- A sensation for inflation: initial swim bladder inflation in larval zebrafish is mediated by the mechanosensory lateral line.The Journal of experimental biology · 2023Article
- A sensation for inflation: initial swim bladder inflation in larval zebrafish is mediated by the mechanosensory lateral line.bioRxiv : the preprint server for biology · 2023Article
- Polarized transport of membrane and secreted proteins during lumen morphogenesis.Seminars in cell & developmental biology · 2023Review
- Morphogenetic Roles of Hydrostatic Pressure in Animal Development.Annual review of cell and developmental biology · 2022Review
- Knockout ofBiomedicines · 2021Article
- Origami: Single-cell 3D shape dynamics oriented along the apico-basal axis of folding epithelia from fluorescence microscopy data.PLoS computational biology · 2021Article
- Fxr signaling and microbial metabolism of bile salts in the zebrafish intestine.Science advances · 2021Article
- TET1-mediated DNA hydroxymethylation regulates adult remyelination in mice.Nature communications · 2021Article
- How Zebrafish Can Drive the Future of Genetic-based Hearing and Balance Research.Journal of the Association for Research in Otolaryngology : JARO · 2021Review
- Programmed and self-organized flow of information during morphogenesis.Nature reviews. Molecular cell biology · 2021Review
- Phenobarbital, midazolam, bumetanide, and neonatal seizures: The devil is in the details.Epilepsia · 2021Article
- In Vivo Cochlear imaging provides a tool to study endolymphatic hydrops.Journal of vestibular research : equilibrium & orientation · 2021Article
- Shedding new light on early sex determination in zebrafish.Archives of toxicology · 2020Article
- SLC12A2 variants cause a neurodevelopmental disorder or cochleovestibular defect.Brain : a journal of neurology · 2020Article
- Neuronal activity disrupts myelinated axon integrity in the absence of NKCC1b.The Journal of cell biology · 2020Article
- Immunological characterization of two types of ionocytes in the inner ear epithelium of Pacific Chub Mackerel (Scomber japonicus).Journal of comparative physiology. B, Biochemical, systemic, and environmental physiology · 2020Article
- Variants encoding a restricted carboxy-terminal domain of SLC12A2 cause hereditary hearing loss in humans.PLoS genetics · 2020Article
- Adult and Developing Zebrafish as Suitable Models for Cardiac Electrophysiology and Pathology in Research and Industry.Frontiers in physiology · 2020Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
2 authors at 1 institution in 1 country.
Funding
Abstract
Endolymph is the specialised extracellular fluid present inside the inner ear. In mammals, disruptions to endolymph homeostasis can result in either collapse or distension of the endolymphatic compartment in the cochlea, with concomitant hearing loss. The zebrafish little ears (lte) mutant shows a collapse of the otic vesicle in the larva, apparently owing to a loss of endolymphatic fluid in the ear, together with an over-inflation of the swim bladder. Mutant larvae display signs of abnormal vestibular function by circling and swimming upside down. The two available alleles of lte are homozygous lethal: mutant larvae fail to thrive beyond 6 days post-fertilisation. Patterning of the otic vesicle is apparently normal. However, the expression of several genes thought to play a role in endolymph production is downregulated, including the sodium-potassium-chloride cotransporter gene nkcc1 (slc12a2) and several Na(+)/K(+)-ATPase channel subunit genes. We show here that lte mutations correspond to lesions in nkcc1. Each allele has a point mutation that disrupts splicing, leading to frame shifts in the coding region that predict the generation of truncated products. Endolymph collapse in the lte/nkcc1 mutant shows distinct parallels to that seen in mouse Nkcc1 mutants, validating zebrafish as a model for the study of endolymph disorders. The collapse in ear volume can be ameliorated in the to27d allele of lte by injection of a morpholino that blocks splicing at an ectopic site introduced by the mutation. This exemplifies the use of morpholinos as potential therapeutic agents for genetic disease.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
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.