نتایج جستجو برای: pick type c1 npc1

تعداد نتایج: 1363808  

Journal: :The Journal of biological chemistry 2012
Miao Xu Ke Liu Manju Swaroop Forbes D Porter Rohini Sidhu Sally Firnkes Daniel S Ory Juan J Marugan Jingbo Xiao Noel Southall William J Pavan Cristin Davidson Steven U Walkley Alan T Remaley Ulrich Baxa Wei Sun John C McKew Christopher P Austin Wei Zheng

Niemann-Pick disease type C (NPC) and Wolman disease are two members of a family of storage disorders caused by mutations of genes encoding lysosomal proteins. Deficiency in function of either the NPC1 or NPC2 protein in NPC disease or lysosomal acid lipase in Wolman disease results in defective cellular cholesterol trafficking. Lysosomal accumulation of cholesterol and enlarged lysosomes are s...

Journal: :Open Access Macedonian Journal of Medical Sciences 2020

Journal: :Current Biology 2001
Mark Zervas Kyra L Somers Mary Anna Thrall Steven U Walkley

Niemann-Pick type C (NPC) disease is a cholesterol lipidosis caused by mutations in NPC1 and NPC2 gene loci. Most human cases are caused by defects in NPC1, as are the spontaneously occurring NPC diseases in mice and cats. NPC1 protein possesses a sterol-sensing domain and has been localized to vesicles that are believed to facilitate the recycling of unesterified cholesterol from late endosome...

Journal: :The Journal of biological chemistry 2007
Agnes Kulinski Jean E Vance

Niemann-Pick C (NPC) disease is a fatal inherited disorder characterized by an accumulation of cholesterol and other lipids in late endosomes/lysosomes. Although this disease is considered to be primarily a neurodegenerative disorder, many NPC patients suffer from liver disease. We have investigated alterations that occur in hepatic lipid homeostasis using primary hepatocytes isolated from NPC1...

Journal: :American journal of physiology. Lung cellular and molecular physiology 2012
Blair R Roszell Jian-Qin Tao Kevin J Yu Shaohui Huang Sandra R Bates

The Niemann-Pick C (NPC) pathway plays an essential role in the intracellular trafficking of cholesterol by facilitating the release of lipoprotein-derived sterol from the lumen of lysosomes. Regulation of cellular cholesterol homeostasis is of particular importance to lung alveolar type II cells because of the need for production of surfactant with an appropriate lipid composition. We performe...

Journal: :Journal of virology 2009
Yuyang Tang Ihid Carneiro Leao Ebony M Coleman Robin Shepard Broughton James E K Hildreth

Human immunodeficiency virus type 1 (HIV-1) relies on cholesterol-laden lipid raft membrane microdomains for entry into and egress out of susceptible cells. In the present study, we examine the need for intracellular cholesterol trafficking pathways with respect to HIV-1 biogenesis using Niemann-Pick type C-1 (NPC1)-deficient (NPCD) cells, wherein these pathways are severely compromised, causin...

Journal: :Molecular & cellular proteomics : MCP 2015
Navin Rauniyar Kanagaraj Subramanian Mathieu Lavallée-Adam Salvador Martínez-Bartolomé William E Balch John R Yates

Niemann-Pick type C (NPC) disease is a fatal neurodegenerative disorder characterized by the accumulation of unesterified cholesterol in the late endosomal/lysosomal compartments. Mutations in the NPC1 protein are implicated in 95% of patients with NPC disease. The most prevalent mutation is the missense mutation I1061T that occurs in ∼ 15-20% of the disease alleles. In our study, an isobaric l...

Journal: :Journal of Neurology, Neurosurgery & Psychiatry 2005

Journal: :Journal of lipid research 2007
Eduardo P Beltroy Benny Liu John M Dietschy Stephen D Turley

Niemann-Pick type C (NPC) disease is a multisystem disorder resulting from mutations in the NPC1 gene that encodes a protein involved in intracellular cholesterol trafficking. Significant liver dysfunction is frequently seen in patients with this disease. The current studies used npc1 mutant mice to investigate the association between liver dysfunction and unesterified cholesterol accumulation,...

2016
Anna Brown Siddharth Patel Carl Ward Anna Lorenz Mauren Ortiz Allison DuRoss Fabian Wieghardt Amanda Esch Elsje G. Otten Laura M. Heiser Viktor I. Korolchuk Conroy Sun Sovan Sarkar Gaurav Sahay

2-Hydroxy-propyl-β-cyclodextrin (HPβCD), a cholesterol scavenger, is currently undergoing Phase 2b/3 clinical trial for treatment of Niemann Pick Type C-1 (NPC1), a fatal neurodegenerative disorder that stems from abnormal cholesterol accumulation in the endo/lysosomes. Unfortunately, the extremely high doses of HPβCD required to prevent progressive neurodegeneration exacerbates ototoxicity, pu...

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