ArticleJournal of cell science2015
Regulation of melanosome number, shape and movement in the zebrafish retinal pigment epithelium by OA1 and PMEL.
Article in Journal of cell science, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.
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Who cites it
31 citing papers in PubMed, 59 citations in OpenAlex.
- Article
- L-DOPA enhances iRPE differentiation via Wnt signaling and improves cytotherapy for retinal degradation.Stem cell research & therapy · 2026Article
- Retinal Vulnerability to Light Pollution in Vitiligo: A Narrative Review of the Role of Melanin Loss in Ocular Phototoxicity.Dermatology (Basel, Switzerland) · 2026Review
- Effect of dual optical pulse scheme with supra and subthreshold fluences on laser micro ablation of pigment.Scientific reports · 2025Article
- The Impact of Photopigment Bleaching on the Human Rod Photoreceptor Subretinal Space Measured Via Optical Coherence Tomography.Investigative ophthalmology & visual science · 2024Article
- Identification of Candidate Genes for Red-Eyed (Albinism) Domestic Guppies Using Genomic and Transcriptomic Analyses.International journal of molecular sciences · 2024Article
- Disrupting the Repeat Domain of Premelanosome Protein (PMEL) Produces Dysamyloidosis and Dystrophic Ocular Pigment Reflective of Pigmentary Glaucoma.International journal of molecular sciences · 2023Article
- New insights into the retinal microstructure-diurnal activity relationship in the African five-lined skink (Trachylepis quinquetaeniata) (Lichtenstein, 1823).Zoological letters · 2023Article
- Structural analysis of melanosomes in living mammalian cells using scanning electron-assisted dielectric microscopy with deep neural network.Computational and structural biotechnology journal · 2023Article
- Transcriptome Analysis Reveals the Complex Regulatory Pathway of Background Color in JuvenileInternational journal of molecular sciences · 2022Article
- CRISPR-AsCas12a Efficiently Corrects aThe CRISPR journal · 2022Article
- Microtubule Motor Transport of Organelles in a Specialized Epithelium: The RPE.Frontiers in cell and developmental biology · 2022Review
- Identification of Long Noncoding RNAs Involved in Eyelid Pigmentation of Hereford Cattle.Frontiers in genetics · 2022Article
- Effect of NK-5962 on Gene Expression Profiling of Retina in a Rat Model of Retinitis Pigmentosa.International journal of molecular sciences · 2021Article
- Melanosome Biogenesis in the Pigmentation of Mammalian Skin.Integrative and comparative biology · 2021Review
- Identification of a novel GPR143 mutation in a large Chinese family with isolated foveal hypoplasia.BMC ophthalmology · 2021Article
- Organelle size scaling over embryonic development.Wiley interdisciplinary reviews. Developmental biology · 2020Review
- Microtubule motor transport in the delivery of melanosomes to the actin-rich apical domain of the retinal pigment epithelium.Journal of cell science · 2020Article
- The cell biology of the retinal pigment epithelium.Progress in retinal and eye research · 2020Review
- The Cytoskeleton of the Retinal Pigment Epithelium: from Normal Aging to Age-Related Macular Degeneration.International journal of molecular sciences · 2019Review
Corrections and comments
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Authors and funding
5 authors at 2 institutions in 1 country.
Funding
Abstract
Analysis of melanosome biogenesis in the retinal pigment epithelium (RPE) is challenging because it occurs predominantly in a short embryonic time window. Here, we show that the zebrafish provides an ideal model system for studying this process because in the RPE the timing of melanosome biogenesis facilitates molecular manipulation using morpholinos. Morpholino-mediated knockdown of OA1 (also known as GPR143), mutations in the human homologue of which cause the most common form of human ocular albinism, induces a major reduction in melanosome number, recapitulating a key feature of the mammalian disease where reduced melanosome numbers precede macromelanosome formation. We further show that PMEL, a key component of mammalian melanosome biogenesis, is required for the generation of cylindrical melanosomes in zebrafish, which in turn is required for melanosome movement into the apical processes and maintenance of photoreceptor integrity. Spherical and cylindrical melanosomes containing similar melanin volumes co-exist in the cell body but only cylindrical melanosomes enter the apical processes. Taken together, our findings indicate that melanosome number and shape are independently regulated and that melanosome shape controls a function in the RPE that depends on localisation in the apical processes.
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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.