نتایج جستجو برای: npc1

تعداد نتایج: 544  

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2004
David E Sleat Jennifer A Wiseman Mukarram El-Banna Sandy M Price Lucie Verot Michael M Shen G Stephen Tint Marie T Vanier Steven U Walkley Peter Lobel

Niemann-Pick C (NPC) disease is a fatal neurodegenerative disorder characterized by a lysosomal accumulation of cholesterol and other lipids within the cells of patients. Clinically identical forms of NPC disease are caused by defects in either of two different proteins: NPC1, a lysosomal-endosomal transmembrane protein and NPC2, a soluble lysosomal protein with cholesterol binding properties. ...

Journal: :Journal of lipid research 2010
William S Garver David Jelinek F John Meaney James Flynn Kathleen M Pettit Glen Shepherd Randall A Heidenreich Cate M Walsh Vockley Graciela Castro Gordon A Francis

Niemann-Pick type C1 disease (NPC1) is an autosomal recessive lysosomal storage disorder characterized by neonatal jaundice, hepatosplenomegaly, and progressive neurodegeneration. The present study provides the lipid profiles, mutations, and corresponding associations with the biochemical phenotype obtained from NPC1 patients who participated in the National NPC1 Disease Database. Lipid profile...

Journal: :eLife 2015
Melinda Ng Esther Ndungo Maria E Kaczmarek Andrew S Herbert Tabea Binger Ana I Kuehne Rohit K Jangra John A Hawkins Robert J Gifford Rohan Biswas Ann Demogines Rebekah M James Meng Yu Thijn R Brummelkamp Christian Drosten Lin-Fa Wang Jens H Kuhn Marcel A Müller John M Dye Sara L Sawyer Kartik Chandran

Biological factors that influence the host range and spillover of Ebola virus (EBOV) and other filoviruses remain enigmatic. While filoviruses infect diverse mammalian cell lines, we report that cells from African straw-colored fruit bats (Eidolon helvum) are refractory to EBOV infection. This could be explained by a single amino acid change in the filovirus receptor, NPC1, which greatly reduce...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2016
Xiaochun Li Jiawei Wang Elias Coutavas Hang Shi Qi Hao Günter Blobel

Niemann-Pick C1 protein (NPC1) is a late-endosomal membrane protein involved in trafficking of LDL-derived cholesterol, Niemann-Pick disease type C, and Ebola virus infection. NPC1 contains 13 transmembrane segments (TMs), five of which are thought to represent a "sterol-sensing domain" (SSD). Although present also in other key regulatory proteins of cholesterol biosynthesis, uptake, and signal...

Journal: :Human molecular genetics 2013
Sophie Louwette Luc Régal Christine Wittevrongel Chantal Thys Gwenny Vandeweeghde Elisa Decuyper Peter Leemans Rita De Vos Chris Van Geet Jaak Jaeken Kathleen Freson

Niemann-Pick type C is a lysosomal storage disease associated with mutations in NPC1 or NPC2, resulting in an accumulation of cholesterol in the endosomal-lysosomal system. Niemann-Pick type C has a clinical spectrum that ranges from a neonatal rapidly fatal disorder to an adult-onset chronic neurodegenerative disease combined with remarkably, in some cases, hematological defects such as thromb...

2013
Blair R. Roszell Jian-Qin Tao Kevin J. Yu Ling Gao Shaohui Huang Yue Ning Sheldon I. Feinstein Charles H. Vite Sandra R. Bates

Niemann-Pick C (NPC) disease is due to loss of NPC1 or NPC2 protein function that is required for unesterified cholesterol transport from the endosomal/lysosomal compartment. Though lung involvement is a recognized characteristic of Niemann-Pick type C disease, the pathological features are not well understood. We investigated components of the surfactant system in both NPC1 mutant mice and fel...

2017
Adriana Lo Castro Michela Murdocca Sabina Pucci Anna Zaratti Chiara Greggi Federica Sangiuolo Virginia Tancredi Claudio Frank Giovanna D’Arcangelo

Niemann-Pick type C disease (NPCD) is an autosomal recessive storage disorder, characterized by abnormal sequestration of unesterified cholesterol within the late endo-lysosomal compartment of cells. In the central nervous system, hypoxic insults could result in low-density lipoprotein (LDL) oxidation and Lectin-like oxidized LDL receptor-1 (LOX-1) induction, leading to a pathological hippocamp...

2012
Bjoern von Einem Petra Weber Michael Wagner Martina Malnar Marko Kosicek Silva Hecimovic Christine A. F. von Arnim Herbert Schneckenburger

Förster resonance energy transfer (FRET) -based techniques have recently been applied to study the interactions between β-site APP-cleaving enzyme-GFP (BACE1-GFP) and amyloid precursor protein-mRFP (APP-mRFP) in U373 glioblastoma cells. In this context, the role of APP-BACE1 proximity in Alzheimer's disease (AD) pathogenesis has been discussed. FRET was found to depend on intracellular choleste...

Journal: :Journal of lipid research 2010
Mark Charman Barry E Kennedy Nolan Osborne Barbara Karten

Niemann-Pick Type C (NPC) disease is a fatal, neurodegenerative disorder, caused in most cases by mutations in the late endosomal protein NPC1. A hallmark of NPC disease is endosomal cholesterol accumulation and an impaired cholesterol homeostatic response, which might affect cholesterol transport to mitochondria and, thus, mitochondrial and cellular function. This study aimed to characterize m...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2008
Rodney E Infante Michael L Wang Arun Radhakrishnan Hyock Joo Kwon Michael S Brown Joseph L Goldstein

Egress of lipoprotein-derived cholesterol from lysosomes requires two lysosomal proteins, polytopic membrane-bound Niemann-Pick C1 (NPC1) and soluble Niemann-Pick C2 (NPC2). The reason for this dual requirement is unknown. Previously, we showed that the soluble luminal N-terminal domain (NTD) of NPC1 (amino acids 25-264) binds cholesterol. This NTD is designated NPC1(NTD). We and others showed ...

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