Regulation of melanosome number, shape and movement in the zebrafish retinal pigment epithelium by OA1 and PMEL
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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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In vertebrate eyes, cylindrically-shaped melanosomes enter the narrow apical processes of the RPE and surround the OSs with their long axis parallel to the OSs and the direction of incoming light.
“In vertebrate eyes, cylindrically-shaped melanosomes enter the narrow apical processes of the RPE, and surround the OSs, with their long axis parallel to the OSs, and thus the direction of incoming light (Troutt and Burnside, 1989; Burgoyne et al., 2015).”
From The cell biology of the retinal pigment epithelium (2020) · cites this paper as Burgoyne et al., 2015
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- Authors
- Burgoyne, T., O'Connor, M. N., Seabra, M. C., Cutler, D. F., & Futter, C. E.
- Affiliations
- University College London (GB)
- MRC Laboratory for Molecular Cell Biology (GB)
- Imperial College London (GB)
- Corresponding author
- Burgoyne, Thomas
- Futter, Clare E.
- Venue
- Journal of Cell Science
- Publisher
- The Company of Biologists
- Published
- 2015-04-01
- Volume
- 128
- Issue
- 7
- Pages
- 1400-1407
- ISSN
- 0021-9533 (ISSN-L)
- 1477-9137
- PMC
- PMC4379728
- OpenAlex
- W2068843391
- Cited by
- 68
- References
- 41
- Citations by year
- 2026: 9 · 2025: 7 · 2024: 4 · 2023: 4 · 2022: 10 · 2021: 10 · 2020: 7 · 2019: 5 · 2018: 4 · 2017: 4 · 2016: 3 · 2015: 1
- FWCI
- 2.13
- Type
- journal article
- Language
- en
- Open access
- hybrid
- doi.org
- Locations
- Journal of Cell Science (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- UCL Discovery (University College London) (submittedVersion)
- Funders
- Wellcome Trust (093445)
- Medical Research Council (MC_UU_12018/2)
- Biotechnology and Biological Sciences Research Council (BB/D011841/1, BB/D011841)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › melanin and skin pigmentation
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Retinal Development and Disorders
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › Zebrafish Biomedical Research Applications
- Keywords
- melanosome biogenesis, retinal pigment epithelium, zebrafish, GPR143, PMEL, ocular albinism, apical processes, photoreceptor integrity, morpholino knockdown
- MeSH terms
- Animals, Disease Models, Animal, Humans, Zebrafish, Albinism, Ocular, Melanosomes, Zebrafish Proteins, Receptors, G-Protein-Coupled, Retinal Pigment Epithelium
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