ArticleeLife2016
The E3 ligase Ubr3 regulates Usher syndrome and MYH9 disorder proteins in the auditory organs of Drosophila and mammals.
Article in eLife, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed, 32 citations in OpenAlex.
- The ubiquitin-proteasome system and proteostasis in the cochlea and sensorineural hearing disorders.Molecular biomedicine · 2026Review
- The deubiquitinase USP17LA negatively regulates T-cell activation and attenuates anti-tumor immunity.EMBO reports · 2025Article
- A role for Myo-II zipper and spaghetti squash in Gliotactin-dependent Drosophila melanogaster wing hair planar cell polarity.PloS one · 2025Article
- Comprehensive insights into the impact of bacterial indole-3-acetic acid on sensory preferences in Drosophila melanogaster.Scientific reports · 2024Article
- Article
- Coordinated Regulation of Myonuclear DNA Methylation, mRNA, and miRNA Levels Associates With the Metabolic Response to Rapid Synergist Ablation-Induced Skeletal Muscle Hypertrophy in Female Mice.Function (Oxford, England) · 2024Article
- The cochlea is built to last a lifetime.Hearing research · 2023Review
- The Voltage-Gated Sodium Channel ineNeuro · 2023Article
- UBR3 promotes inflammation and apoptosis via DUSP1/p38 pathway in the nucleus pulposus cells of patients with intervertebral disc degeneration.Human cell · 2022Article
- Genetics, pathogenesis and therapeutic developments for Usher syndrome type 2.Human genetics · 2022Review
- The Drosophila anterior-posterior axis is polarized by asymmetric myosin activation.Current biology : CB · 2022Article
- Article
- Cellular defects resulting from disease-related myosin II mutations inProceedings of the National Academy of Sciences of the United States of America · 2019Article
- Mucin 17 inhibits the progression of human gastric cancer by limiting inflammatory responses through a MYH9-p53-RhoA regulatory feedback loop.Journal of experimental & clinical cancer research : CR · 2019Article
- Dysfunction ofProceedings of the National Academy of Sciences of the United States of America · 2019Article
- Grxcr1 Promotes Hair Bundle Development by Destabilizing the Physical Interaction between Harmonin and Sans Usher Syndrome Proteins.Cell reports · 2018Article
- Stepwise wiring of theeLife · 2018Article
- Pathways to neurodegeneration: lessons learnt from unbiased genetic screens inJournal of genetics · 2018Review
- UsingDisease models & mechanisms · 2018Review
- Hearing lessons from flies.eLife · 2016Article
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Authors and funding
8 authors at 4 institutions in 2 countries.
Funding
Abstract
Myosins play essential roles in the development and function of auditory organs and multiple myosin genes are associated with hereditary forms of deafness. Using a forward genetic screen in Drosophila, we identified an E3 ligase, Ubr3, as an essential gene for auditory organ development. Ubr3 negatively regulates the mono-ubiquitination of non-muscle Myosin II, a protein associated with hearing loss in humans. The mono-ubiquitination of Myosin II promotes its physical interaction with Myosin VIIa, a protein responsible for Usher syndrome type IB. We show that ubr3 mutants phenocopy pathogenic variants of Myosin II and that Ubr3 interacts genetically and physically with three Usher syndrome proteins. The interactions between Myosin VIIa and Myosin IIa are conserved in the mammalian cochlea and in human retinal pigment epithelium cells. Our work reveals a novel mechanism that regulates protein complexes affected in two forms of syndromic deafness and suggests a molecular function for Myosin IIa in auditory organs.
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