نتایج جستجو برای: pick type c

تعداد نتایج: 2248990  

2017
Alfred L Yergey Paul S Blank Stephanie M Cologna Peter S Backlund Forbes D Porter Allan J Darling

2-Hydroxypropyl-beta-cyclodextrin (HPβCD) has gained recent attention as a potential therapeutic intervention in the treatment of the rare autosomal-recessive, neurodegenerative lysosomal storage disorder Niemann-Pick Disease Type C1 (NPC1). Notably, HPβCD formulations are not comprised of a single molecular species, but instead are complex mixtures of species with differing degrees of hydroxyp...

Journal: :Journal of Neurology, Neurosurgery & Psychiatry 2005

2011
Sayali S. Dixit Michel Jadot Istvan Sohar David E. Sleat Ann M. Stock Peter Lobel

Niemann-Pick Type C (NPC) disease is a lysosomal storage disorder characterized by accumulation of unesterified cholesterol and other lipids in the endolysosomal system. NPC disease results from a defect in either of two distinct cholesterol-binding proteins: a transmembrane protein, NPC1, and a small soluble protein, NPC2. NPC1 and NPC2 are thought to function closely in the export of lysosoma...

Journal: :Cell 2009
Hyock Joo Kwon Lina Abi-Mosleh Michael L. Wang Johann Deisenhofer Joseph L. Goldstein Michael S. Brown Rodney E. Infante

LDL delivers cholesterol to lysosomes by receptor-mediated endocytosis. Exit of cholesterol from lysosomes requires two proteins, membrane-bound Niemann-Pick C1 (NPC1) and soluble NPC2. NPC2 binds cholesterol with its isooctyl side chain buried and its 3beta-hydroxyl exposed. Here, we describe high-resolution structures of the N-terminal domain (NTD) of NPC1 and complexes with cholesterol and 2...

2016
André R. A. Marques Tanit L. Gabriel Jan Aten Cindy P. A. A. van Roomen Roelof Ottenhoff Nike Claessen Pilar Alfonso Pilar Irún Pilar Giraldo Johannes M. F. G. Aerts Marco van Eijk Andrea Dardis

Impaired function of NPC1 or NPC2 lysosomal proteins leads to the intracellular accumulation of unesterified cholesterol, the primary defect underlying Niemann-Pick type C (NPC) disease. In addition, glycosphingolipids (GSLs) accumulate in lysosomes as well. Intralysosomal lipid accumulation triggers the activation of a set of genes, including potential biomarkers. Transcript levels of Gpnmb ha...

Journal: :Chemistry & biology 2013
Kenji Ohgane Fumika Karaki Kosuke Dodo Yuichi Hashimoto

Niemann-Pick type C1 (NPC1) is a polytopic endosomal membrane protein required for efflux of LDL-derived cholesterol from endosomes, and mutations of this protein are associated with Niemann-Pick disease type C, a fatal neurodegenerative disease. At least one prevalent mutation (I1061T) has been shown to cause a folding defect, which results in failure of endosomal localization, leading to a lo...

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