ArticleHearing research2008
A mouse model with postnatal endolymphatic hydrops and hearing loss.
Article in Hearing research, 2008. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 27 papers.
What it found
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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.
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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.
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Who cites it
27 citing papers in PubMed, 54 citations in OpenAlex.
- Genetic contributions to familial Ménière's disease: a systematic review.European archives of oto-rhino-laryngology : official journal of the European Federation of Oto-Rhino-Laryngological Societies (EUFOS) : affiliated with the German Society for Oto-Rhino-Laryngology - Head and Neck Surgery · 2026Review
- From Anatomy to Application: A Comprehensive Review of Nanovesicular Systems for Otic Drug Delivery.AAPS PharmSciTech · 2026Review
- Hearing impairment in X-linked hypophosphatemia: a review.JBMR plus · 2025Article
- X-Linked Hypophosphataemia and Burosumab: A Systemic Disease With a New Treatment.Journal of paediatrics and child health · 2025Review
- Disease Manifestations and Complications in Dutch X-Linked Hypophosphatemia Patients.Calcified tissue international · 2024Article
- PHEXGenes · 2022Article
- Performance and self-perceived hearing impairment after cochlear implantation in Menière's disease.Laryngoscope investigative otolaryngology · 2022Article
- Conductive Hearing Loss in the Hyp Mouse Model of X-Linked Hypophosphatemia Is Accompanied by Hypomineralization of the Auditory Ossicles.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2021Article
- Experimental Animal Models for Meniere's Disease: A Mini-Review.Journal of audiology & otology · 2020Review
- FGF23 and its role in X-linked hypophosphatemia-related morbidity.Orphanet journal of rare diseases · 2019Review
- Computer-aided detection and quantification of endolymphatic hydrops within the mouse cochlea in vivo using optical coherence tomography.Journal of biomedical optics · 2017Article
- Hypophosphatemic rickets: lessons from disrupted FGF23 control of phosphorus homeostasis.Current osteoporosis reports · 2015Review
- A Genetic Murine Model of Endolymphatic Hydrops: The Phex Mouse.Current otorhinolaryngology reports · 2014Article
- Slc26a4-insufficiency causes fluctuating hearing loss and stria vascularis dysfunction.Neurobiology of disease · 2014Article
- A mouse model validates the utility of electrocochleography in verifying endolymphatic hydrops.Journal of the Association for Research in Otolaryngology : JARO · 2014Article
- Semicircular canal morphogenesis in the zebrafish inner ear requires the function of gpr126 (lauscher), an adhesion class G protein-coupled receptor gene.Development (Cambridge, England) · 2013Article
- What have we learned from murine models of otitis media?Current allergy and asthma reports · 2013Review
- Characterization of neuronal cell death in the spiral ganglia of a mouse model of endolymphatic hydrops.Otology & neurotology : official publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology · 2013Article
- Article
- The expanding family of hypophosphatemic syndromes.Journal of bone and mineral metabolism · 2012Review
Corrections and comments
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Authors and funding
8 authors at 4 institutions in 1 country.
Funding
Abstract
Endolymphatic hydrops (ELH), hearing loss and neuronal degeneration occur together in a variety of clinically significant disorders, including Meniere's disease (MD). However, the sequence of these pathological changes and their relationship to each other are not well understood. In this regard, an animal model that spontaneously develops these features postnatally would be useful for research purposes. A search for such a model led us to the Phex Hyp-Duk mouse, a mutant allele of the Phex gene causing X-linked hypophosphatemic rickets. The hemizygous male (Phex Hyp-Duk/Y) was previously reported to exhibit various abnormalities during adulthood, including thickening of bone, ELH and hearing loss. The reported inner-ear phenotype was suggestive of progressive pathology and spontaneous development of ELH postnatally, but not conclusive. The main focuses of this report are to further characterize the inner ear phenotype in Phex Hyp-Duk/Y mice and to test the hypotheses that (a) the Phex Hyp-Duk/Y mouse develops ELH and hearing loss postnatally, and (b) the development of ELH in the Phex Hyp-Duk/Y mouse is associated with obstruction of the endolymphatic duct (ED) due to thickening of the surrounding bone. Auditory brainstem response (ABR) recordings at various times points and histological analysis of representative temporal bones reveal that Phex Hyp-Duk/Y mice typically develop adult onset, asymmetric, progressive hearing loss closely followed by the onset of ELH. ABR and histological data show that functional degeneration precedes structural degeneration. The major degenerative correlate of hearing loss and ELH in the mutants is the primary loss of spiral ganglion cells. Further, Phex Hyp-Duk/Y mice develop ELH without evidence of ED obstruction, supporting the idea that ELH can be induced by a mechanism other than the blockade of longitudinal flow of endolymphatic fluid, and occlusion of ED is not a prerequisite for the development of ELH in patients.
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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.