{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,4,6]],"date-time":"2025-04-06T22:03:30Z","timestamp":1743977010213},"reference-count":34,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2005,8,16]],"date-time":"2005-08-16T00:00:00Z","timestamp":1124150400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pigment Cell Research"],"published-print":{"date-parts":[[2005,12]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Hermansky\u2013Pudlak syndrome (HPS) is an autosomal recessive disorder resulting from mutations in a family of genes required for efficient transport of lysosomal\u2010related proteins from the trans\u2010Golgi network to a target organelle. To date, there are several genetically distinct forms of HPS. Many forms of HPS exhibit aberrant trafficking of melanosome\u2010targeted proteins resulting in incomplete melanosome biogenesis responsible for oculocutaneous albinism observed in patients. In HPS\u20101, melanosome\u2010targeted proteins are localized to characteristic membranous complexes, which have morphologic similarities to macroautophagosomes. In this report, we evaluated the hypothesis that HPS\u20101\u2010specific membranous complexes comprise a component of the lysosomal compartment of melanocytes. Using indirect immunofluorescence, an increase in co\u2010localization of misrouted tyrosinase with cathepsin\u2010L, a lysosomal cysteine protease, occurred in HPS\u20101 melanocytes. In addition, ribophorin II, an integral endoplasmic reticulum protein that is also a component of macroautophagosomes, and LC3, a specific marker of macrophagosomes, demonstrated localization to membranous complexes in HPS\u20101 melanocytes. At the electron microscopic level, the membranous complexes exhibited acid phosphatase activity and localization of exogenously supplied horseradish peroxidase (HRP)\u2010conjugated gold particles, indicating incorporation of lysosomal and endosomal components to membranous complexes, respectively. These results confirm that membranous complexes of HPS\u20101 melanocytes are macroautophagosomal representatives of the lysosomal compartment.<\/jats:p>","DOI":"10.1111\/j.1600-0749.2005.00265.x","type":"journal-article","created":{"date-parts":[[2010,9,20]],"date-time":"2010-09-20T14:36:35Z","timestamp":1284993395000},"page":"417-426","source":"Crossref","is-referenced-by-count":26,"title":["Membranous complexes characteristic of melanocytes derived from patients with Hermansky\u2013Pudlak syndrome type 1 are macroautophagosomal entities of the lysosomal compartment"],"prefix":"10.1111","volume":"18","author":[{"given":"Justin W.","family":"Smith","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amy","family":"Koshoffer","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Randal E.","family":"Morris","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Raymond E.","family":"Boissy","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2005,8,16]]},"reference":[{"key":"e_1_2_6_2_1","first-page":"1037","article-title":"Altered protein localization in melanocytes from Hermansky\u2013Pudlak syndrome: support for the role of the HPS gene production in intracellular trafficking","volume":"78","author":"Boissy R.E.","year":"1998","journal-title":"Lab. 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