ArticlePLoS genetics2009
Accumulation of rhodopsin in late endosomes triggers photoreceptor cell degeneration.
Article in PLoS genetics, 2009. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 66 papers.
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Who cites it
66 citing papers in PubMed, 99 citations in OpenAlex.
- Enhanced Endocytosis and Mitochondrial Stress Underlie Severe Retinitis Pigmentosa With RHO P347L Mutant.Investigative ophthalmology & visual science · 2026Article
- NECAP antagonizes light-induced Rhodopsin-1 internalization to promote photoreceptor homeostasis.bioRxiv : the preprint server for biology · 2026Article
- PIKfyve preserves endolysosomal function in photoreceptors and RPE cells to maintain retinal integrity.Cell death & disease · 2026Article
- A conventional PKC critical for both the light-dependent and the light-independent regulation of the actin cytoskeleton in Drosophila photoreceptors.The Journal of biological chemistry · 2023Article
- The Role of Reversible Phosphorylation ofInternational journal of molecular sciences · 2022Review
- Review
- Drosophila fabp is required for light-dependent Rhodopsin-1 clearance and photoreceptor survival.PLoS genetics · 2021Article
- Mutations in the splicing regulator Prp31 lead to retinal degeneration inBiology open · 2021Article
- Measurement of Diurnal Variation in Rod Outer Segment Length In Vivo in Mice With the OCT Optoretinogram.Investigative ophthalmology & visual science · 2020Article
- RNA-Seq reveals differential expression profiles and functional annotation of genes involved in retinal degeneration in Pde6c mutant Danio rerio.BMC genomics · 2020Article
- Identification of Genes Required for Apical Protein Trafficking inG3 (Bethesda, Md.) · 2019Article
- Immunocytochemical Labeling of Rhabdomeric Proteins inThe journal of histochemistry and cytochemistry : official journal of the Histochemistry Society · 2019Article
- CaThe Journal of neuroscience : the official journal of the Society for Neuroscience · 2019Article
- Human eye conditions: insights from the fly eye.Human genetics · 2019Review
- Whole Genome Sequencing of Giant Schnauzer Dogs with Progressive Retinal Atrophy EstablishesGenes · 2019Article
- Rab-mediated trafficking in the secondary cells of Drosophila male accessory glands and its role in fecundity.Traffic (Copenhagen, Denmark) · 2019Article
- Changes in endolysosomal organization define a pre-degenerative state in the crumbs mutant Drosophila retina.PloS one · 2019Article
- Regulation of Membrane Turnover by Phosphatidic Acid: Cellular Functions and Disease Implications.Frontiers in cell and developmental biology · 2019Review
- The V-ATPase V1 subunit A1 is required for rhodopsin anterograde trafficking in Drosophila.Molecular biology of the cell · 2018Article
- Live Observation of Two Parallel Membrane Degradation Pathways at Axon Terminals.Current biology : CB · 2018Article
6 more citing papers are in PubMed but not listed here.
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Progressive retinal degeneration is the underlying feature of many human retinal dystrophies. Previous work using Drosophila as a model system and analysis of specific mutations in human rhodopsin have uncovered a connection between rhodopsin endocytosis and retinal degeneration. In these mutants, rhodopsin and its regulatory protein arrestin form stable complexes, and endocytosis of these complexes causes photoreceptor cell death. In this study we show that the internalized rhodopsin is not degraded in the lysosome but instead accumulates in the late endosomes. Using mutants that are defective in late endosome to lysosome trafficking, we were able to show that rhodopsin accumulates in endosomal compartments in these mutants and leads to light-dependent retinal degeneration. Moreover, we also show that in dying photoreceptors the internalized rhodopsin is not degraded but instead shows characteristics of insoluble proteins. Together these data implicate buildup of rhodopsin in the late endosomal system as a novel trigger of death of photoreceptor neurons.
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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.