ArticleProceedings of the National Academy of Sciences of the United States of America2004
dsu functions in a MYO5A-independent pathway to suppress the coat color of dilute mice.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2004. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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Who cites it
25 citing papers in PubMed, 45 citations in OpenAlex.
- Melanoregulin Deficiency Affects Bone Maintenance and Lean Body Mass.bioRxiv : the preprint server for biology · 2025Article
- Comparison of Transcriptome Differences in Scales of Two Closely Related Snake Species (Animals : an open access journal from MDPI · 2025Article
- Population Genomics Reveals Elevated Inbreeding and Accumulation of Deleterious Mutations in White Raccoon Dogs.Biology · 2025Article
- Melanocytes in regenerative medicine applications and disease modeling.Journal of translational medicine · 2024Review
- Genetic Basis of Pigment Dispersion Syndrome and Pigmentary Glaucoma: An Update and Functional Insights.Genes · 2024Review
- E2F1-induced microRNA-224-5p expression is associated with hepatocellular carcinoma cell migration, invasion and epithelial-mesenchymal transition via MREG.Oncology letters · 2022Article
- Melanin Transfer and Fate within Keratinocytes in Human Skin Pigmentation.Integrative and comparative biology · 2021Article
- Mapping of Adaptive Traits Enabled by a High-Density Linkage Map for Lake Trout.G3 (Bethesda, Md.) · 2020Article
- Four Novel SNPs ofAnimals : an open access journal from MDPI · 2019Article
- The Structure of Melanoregulin Reveals a Role for Cholesterol Recognition in the Protein's Ability to Promote Dynein Function.Structure (London, England : 1993) · 2018Article
- Molecular Genetics of Pigment Dispersion Syndrome and Pigmentary Glaucoma: New Insights into Mechanisms.Journal of ophthalmology · 2018Review
- Phagocytosis-dependent ketogenesis in retinal pigment epithelium.The Journal of biological chemistry · 2017Article
- The Contribution of Melanoregulin to Microtubule-Associated Protein 1 Light Chain 3 (LC3) Associated Phagocytosis in Retinal Pigment Epithelium.Molecular neurobiology · 2015Article
- Melanosome transfer: it is best to give and receive.Current opinion in cell biology · 2014Review
- The GTPase-deficient Rab27A(Q78L) mutant inhibits melanosome transport in melanocytes through trapping of Rab27A effector protein Slac2-a/melanophilin in their cytosol: development of a novel melanosome-targetinG tag.The Journal of biological chemistry · 2014Article
- Loss of melanoregulin (MREG) enhances cathepsin-D secretion by the retinal pigment epithelium.Visual neuroscience · 2013Article
- Melanoregulin is stably targeted to the melanosome membrane by palmitoylation.Biochemical and biophysical research communications · 2012Article
- The Rab interacting lysosomal protein (RILP) homology domain functions as a novel effector domain for small GTPase Rab36: Rab36 regulates retrograde melanosome transport in melanocytes.The Journal of biological chemistry · 2012Article
- Melanoregulin regulates a shedding mechanism that drives melanosome transfer from melanocytes to keratinocytes.Proceedings of the National Academy of Sciences of the United States of America · 2012Article
- Modifier genes and the plasticity of genetic networks in mice.PLoS genetics · 2012Review
Corrections and comments
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Authors and funding
9 authors at 4 institutions in 3 countries.
Funding
Abstract
MYO5A is a major actin-based vesicle transport motor that binds to one of its cargos, the melanosome, by means of a RAB27A/MLPH receptor. When one of the members of this receptor-motor complex is mutated, the melanosomes clump in the perinuclear region of the melanocyte and are transferred unevenly to the developing hair, leading to a dilution of coat color. Mutation of a fourth gene, dilute suppressor (dsu), suppresses this coat color dilution. MYO5A is required for the peripheral accumulation of melanosomes in melanocytes, but its role in melanosome transfer to neighboring keratinocytes and the hair is unknown. Here, we show that MYO5A is nonessential for melanosome transfer, although pigment incorporation into the hair in MYO5A-deficient mice is uneven, probably due to the clumping of melanosomes that occurs in the perinuclear region of mutant melanocytes. We also show that dsu is caused by a loss-of-function mutation in a unique vertebrate-specific protein that appears to function in an MYO5A-independent pathway to alter pigment incorporation into the hair. Therefore, dsu identifies a unique protein involved in pigmentation of the mammalian hair.
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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.