ArticleProceedings of the National Academy of Sciences of the United States of America1995
Mouse model for Usher syndrome: linkage mapping suggests homology to Usher type I reported at human chromosome 11p15.
Article in Proceedings of the National Academy of Sciences of the United States of America, 1995. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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Who cites it
41 citing papers in PubMed, 107 citations in OpenAlex.
- Article
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- Inpp5e is crucial for photoreceptor outer segment maintenance.Journal of cell science · 2025Article
- Abca4, mutated in Stargardt disease, is required for structural integrity of cone outer segments.Disease models & mechanisms · 2025Article
- The predawn dilemma in adeno-associated virus-based gene therapies for hereditary deafness.American journal of stem cells · 2025Review
- Contribution of intraflagellar transport to compartmentalization and maintenance of the photoreceptor cell.Proceedings of the National Academy of Sciences of the United States of America · 2024Article
- Extracellular vesicles highlight many cases of photoreceptor degeneration.Frontiers in molecular neuroscience · 2023Review
- Microvesicle release from inner segments of healthy photoreceptors is a conserved phenomenon in mammalian species.Disease models & mechanisms · 2022Article
- A combined genome-wide association and molecular study of age-related hearing loss in H. sapiens.BMC medicine · 2021Article
- Genome-wide identification of the Tubby-Like Protein (TLPs) family in medicinal model plantPeerJ · 2021Article
- Distinct roles of stereociliary links in the nonlinear sound processing and noise resistance of cochlear outer hair cells.Proceedings of the National Academy of Sciences of the United States of America · 2020Article
- Comprehensive Profiling of Tubby-Like Protein Expression Uncovers Ripening-RelatedInternational journal of molecular sciences · 2020Article
- PRCD is essential for high-fidelity photoreceptor disc formation.Proceedings of the National Academy of Sciences of the United States of America · 2019Article
- Phenotypic characterization of P23H and S334ter rhodopsin transgenic rat models of inherited retinal degeneration.Experimental eye research · 2018Article
- Phosphodiesterase Inhibitors Sildenafil and Vardenafil Reduce Zebrafish Rod Photoreceptor Outer Segment Shedding.Investigative ophthalmology & visual science · 2017Article
- Insights from Genetic Model Systems of Retinal Degeneration: Role of Epsins in Retinal Angiogenesis and VEGFR2 Signaling.Journal of nature and science · 2017Article
- Genome-Wide Identification and Expression Analysis of the Tubby-Like Protein Family in theFrontiers in plant science · 2016Article
- Genetic modifiers and oligogenic inheritance.Cold Spring Harbor perspectives in medicine · 2015Review
- New insights into the subcellular localization of Tubby-like proteins and their participation in the Arabidopsis-Piriformospora indica interaction.Plant signaling & behavior · 2013Article
- Sustained protection against photoreceptor degeneration in tubby mice by intravitreal injection of nanoceria.Biomaterials · 2012Article
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Authors and funding
7 authors at 4 institutions in 2 countries.
Funding
Abstract
Usher syndrome is a group of diseases with autosomal recessive inheritance, congenital hearing loss, and the development of retinitis pigmentosa, a progressive retinal degeneration characterized by night blindness and visual field loss over several decades. The causes of Usher syndrome are unknown and no animal models have been available for study. Four human gene sites have been reported, suggesting at least four separate forms of Usher syndrome. We report a mouse model of type I Usher syndrome, rd5, whose linkage on mouse chromosome 7 to Hbb and tub has homology to human Usher I reported on human chromosome 11p15. The electroretinogram in homozygous rd5/rd5 mouse is never normal with reduced amplitudes that extinguish by 6 months. Auditory-evoked response testing demonstrates increased hearing thresholds more than control at 3 weeks of about 30 decibels (dB) that worsen to about 45 dB by 6 months.
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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.