ArticleThe American journal of pathology2005
Melanocyte-specific proteins are aberrantly trafficked in melanocytes of Hermansky-Pudlak syndrome-type 3.
Article in The American journal of pathology, 2005. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 45 citations in OpenAlex.
- Dermatologic manifestations in patients with the Hermansky-Pudlak syndrome types 1 and 3.Orphanet journal of rare diseases · 2022Article
- Hermansky-Pudlak syndrome: Mutation update.Human mutation · 2020Article
- The road to lysosome-related organelles: Insights from Hermansky-Pudlak syndrome and other rare diseases.Traffic (Copenhagen, Denmark) · 2019Review
- BLOC-2 targets recycling endosomal tubules to melanosomes for cargo delivery.The Journal of cell biology · 2015Article
- Melanocytic galectin-3 is associated with tyrosinase-related protein-1 and pigment biosynthesis.The Journal of investigative dermatology · 2015Article
- Essential role of the molecular chaperone gp96 in regulating melanogenesis.Pigment cell & melanoma research · 2014Article
- Mechanisms of protein delivery to melanosomes in pigment cells.Physiology (Bethesda, Md.) · 2012Review
- Comparison of bone morphogenetic protein-2 and osteoactivin for mesenchymal cell differentiation: effects of bolus and continuous administration.Journal of cellular physiology · 2011Article
- Functional interactions between OCA2 and the protein complexes BLOC-1, BLOC-2, and AP-3 inferred from epistatic analyses of mouse coat pigmentation.Pigment cell & melanoma research · 2011Article
- [Biogenesis of melanosomes - the chessboard of pigmentation].Medecine sciences : M/S · 2011Article
- A case of hermansky-pudlak syndrome with pulmonary sarcoidosis.The Journal of clinical and aesthetic dermatology · 2009Article
- Disorders of lysosome-related organelle biogenesis: clinical and molecular genetics.Annual review of genomics and human genetics · 2008Review
- Melanosomes--dark organelles enlighten endosomal membrane transport.Nature reviews. Molecular cell biology · 2007Review
- Improper trafficking of melanocyte-specific proteins in Hermansky-Pudlak syndrome type-5.The Journal of investigative dermatology · 2007Article
- BLOC-1 is required for cargo-specific sorting from vacuolar early endosomes toward lysosome-related organelles.Molecular biology of the cell · 2007Article
- Regulation of tyrosinase trafficking and processing by presenilins: partial loss of function by familial Alzheimer's disease mutation.Proceedings of the National Academy of Sciences of the United States of America · 2006Article
- Association of the Hermansky-Pudlak syndrome type-3 protein with clathrin.BMC cell biology · 2005Article
- Melanocytes derived from patients with Hermansky-Pudlak Syndrome types 1, 2, and 3 have distinct defects in cargo trafficking.The Journal of investigative dermatology · 2005Article
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
Abstract
Hermansky-Pudlak Syndrome-type 3 (HPS-3) is a relatively mild subtype of HPS with minimal cutaneous and ocular depigmentation. The HPS-3 gene encodes a novel protein of unknown function with a predicted molecular weight of 114 kd. To assess the role of the HPS3 protein in melanization, cultured melanocytes developed from HPS-3 patients were evaluated biochemically and histologically for activity and localization of melanocyte-specific proteins. Endogenous tyrosinase activity of HPS-3 melanocytes was substantial, but tyrosinase activity and melanin synthesis was suppressed in intact melanocytes. However, the level of suppression, as well as extent to which up-regulation by isobutylmethylxanthine and cholera toxin was muted, was less that in HPS-1 melanocytes. Ultrastructurally, HPS-3 melanocytes contained morphologically normal melanosomes, predominantly of stage I and II with minimal stage III and few stage IV melanosomes. Dihydroxyphenylalanine (DOPA) histochemistry demonstrated an increase in melanization of melanosomes. Unique to HPS-3 melanocytes were numerous DOPA-positive 50-nm vesicles and tubular elements present throughout the cell body and dendrites. Tyrosinase, tyrosinase-related protein-1 (Tyrp1), dopachrome tautomerase (Dct), and LAMP1 and 3 localization in HPS-3 melanocytes, as evaluated by immunocytochemistry and confocal microscopy, demonstrated a fine, floccular distribution in contrast to the coarse, granular distribution characteristic of control melanocytes. The localization profile of other proteins expressed by melanocytes (ie, Silver/Pmel17, Melan-A/MART-1, LAMP2, Rab 27, transferrin, c-kit, adaptin-3, and the HPS1 protein) appeared normal. These results suggest that a specific subset of melanocyte proteins are aberrantly trafficked throughout the HPS-3 melanocyte and may be responsible for the reduction in melanin synthesis.
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