نتایج جستجو برای: npc1

تعداد نتایج: 544  

2016
Esther Ndungo Andrew S. Herbert Matthijs Raaben Gregor Obernosterer Rohan Biswas Emily Happy Miller Ariel S. Wirchnianski Jan E. Carette Thijn R. Brummelkamp Sean P. Whelan John M. Dye Kartik Chandran

Filoviruses are the causative agents of an increasing number of disease outbreaks in human populations, including the current unprecedented Ebola virus disease (EVD) outbreak in western Africa. One obstacle to controlling these epidemics is our poor understanding of the host range of filoviruses and their natural reservoirs. Here, we investigated the role of the intracellular filovirus receptor...

Journal: :The Journal of clinical investigation 2008
Jessie R Zhang Trey Coleman S Joshua Langmade David E Scherrer Lindsay Lane M Hunter Lanier Chu Feng Mark S Sands Jean E Schaffer Clay F Semenkovich Daniel S Ory

Niemann-Pick C1 (NPC1) is a key participant in cellular cholesterol trafficking. Loss of NPC1 function leads to defective suppression of SREBP-dependent gene expression and failure to appropriately activate liver X receptor-mediated (LXR-mediated) pathways, ultimately resulting in intracellular cholesterol accumulation. To determine whether NPC1 contributes to regulation of macrophage sterol ho...

Journal: :Journal of lipid research 2006
Barbara Karten Robert B Campenot Dennis E Vance Jean E Vance

Niemann-Pick type C (NPC) disease is a fatal, neurodegenerative disorder caused in 95% of cases by loss of function of NPC1, a ubiquitous endosomal transmembrane protein. A biochemical hallmark of NPC deficiency is cholesterol accumulation in the endocytic pathway. Although cholesterol trafficking defects are observed in all cell types, neurons are the most vulnerable to NPC1 deficiency, sugges...

Journal: :Journal of lipid research 2011
L Ulatowski R Parker C Davidson N Yanjanin T J Kelley D Corey J Atkinson F Porter H Arai S U Walkley D Manor

Vitamin E (α-tocopherol) is the major lipid-soluble antioxidant in many species. Niemann-Pick type C (NPC) disease is a lysosomal storage disorder caused by mutations in the NPC1 or NPC2 gene, which regulates lipid transport through the endocytic pathway. NPC disease is characterized by massive intracellular accumulation of unesterified cholesterol and other lipids in lysosomal vesicles. We exa...

2017
Bichao Zhang Ciqing Yang Liang Qiao Qiuling Li Congrui Wang Xin Yan Juntang Lin

Niemann-Pick disease, type C1 (Npc1), is an atypical lysosomal storage disorder caused by autosomal recessive inheritance of mutations in Npc1 gene. In the Npc1 mutant mice (Npc1-/- ), the initial manifestation is enlarged spleen, concomitant with free cholesterol accumulation. Telocytes (TCs), a novel type of interstitial cell, exist in a variety of tissues including spleen, presumably thought...

Journal: :The Journal of biological chemistry 2005
Wenxin Yu Jian-Sheng Gong Mihee Ko William S Garver Katsuhiko Yanagisawa Makoto Michikawa

Niemann-Pick type C1 (NPC1) disease is a fatal hereditary disorder characterized by a defect in cholesterol trafficking and progressive neurodegeneration. Although the NPC1 gene has been identified, the molecular mechanism responsible for neuronal dysfunction in brains of patients with NPC1 disease remains unknown. This study demonstrates that the amount of cholesterol within mitochondria membr...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2011
Xuefen Xie Michael S Brown John M Shelton James A Richardson Joseph L Goldstein Guosheng Liang

Substitution mutations in adjacent amino acids of the N-terminal domain of NPC1, a lysosomal membrane protein, abolish its cholesterol binding activity and impair its ability to export cholesterol from lysosomes of cultured cells lacking npc1 [Kwon HJ, et al. (2009) Cell 137:1213-1224]. Here, we show that the same two mutations (proline-202 and phenylalanine-203, both changed to alanine) reprod...

2017
Franziska Peter Sebastian Rost Arndt Rolfs Moritz J Frech

BACKGROUND Niemann-Pick disease Type C1 (NPC1) is a rare progressive neurodegenerative disorder caused by mutations in the NPC1 gene. The pathological mechanisms, underlying NPC1 are not yet completely understood. Especially the contribution of glial cells and gliosis to the progression of NPC1, are controversially discussed. As an analysis of affected cells is unfeasible in NPC1-patients, we r...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2004
Nobutaka Ohgami Dennis C Ko Matthew Thomas Matthew P Scott Catherine C Y Chang Ta-Yuan Chang

Niemann-Pick type C (NPC) 1 protein plays important roles in moving cholesterol and other lipids out of late endosomes by means of vesicular trafficking, but it is not known whether NPC1 directly interacts with cholesterol. We performed photoaffinity labeling of intact cells expressing fluorescent protein (FP)-tagged NPC1 by using [(3)H]7,7-azocholestanol ([(3)H]AC). After immunoprecipitation, ...

2017
Tobias Schwerd Sumeet Pandey Huei-Ting Yang Katrin Bagola Elisabeth Jameson Jonathan Jung Robin H Lachmann Neil Shah Smita Y Patel Claire Booth Heiko Runz Gesche Düker Ruth Bettels Marianne Rohrbach Subra Kugathasan Helen Chapel Satish Keshav Abdul Elkadri Nick Platt Alexio M Muise Sibylle Koletzko Ramnik J Xavier Thorsten Marquardt Fiona Powrie James E Wraith Mads Gyrd-Hansen Frances M Platt Holm H Uhlig

OBJECTIVE Patients with Niemann-Pick disease type C1 (NPC1), a lysosomal lipid storage disorder that causes neurodegeneration and liver damage, can present with IBD, but neither the significance nor the functional mechanism of this association is clear. We studied bacterial handling and antibacterial autophagy in patients with NPC1. DESIGN We characterised intestinal inflammation in 14 patien...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید