نتایج جستجو برای: pick type c1 npc1

تعداد نتایج: 1363808  

Journal: :Human molecular genetics 2011
David Jelinek Veronica Millward Amandip Birdi Theodore P Trouard Randall A Heidenreich William S Garver

A recent population-based genome-wide association study has revealed that the Niemann-Pick C1 (NPC1) gene is associated with early-onset and morbid adult obesity. Concurrently, our candidate gene-based mouse growth study performed using the BALB/cJ NPC1 mouse model (Npc1) with decreased Npc1 gene dosage independently supported these results by suggesting an Npc1 gene-diet interaction in relatio...

2013
Farshad Tamari Fannie W. Chen Chunlei Li Jagrutiben Chaudhari Yiannis A. Ioannou

Activation of protein kinase C (PKC) has previously been shown to ameliorate the cholesterol transport defect in Niemann Pick Type C1 (NPC1) cells, presumably by increasing the soluble levels of one of its substrates, vimentin. This activity would then restore the vimentin cycle in these cells and allow vimentin-dependent retrograde transport to proceed. Here, we further investigate the effects...

2010
Forbes D. Porter

tables on cholesterol by using its oxidation products to diagnose and treat the disease in its earliest stages. drug efficacy. Free cholesterol may be at the root of Niemann-Pick C1 disease, but now, there is a way to turn the markers of early clinical disease and can be used not only to monitor disease progression but also to demonstrate or diabetes. Together, these compelling results suggest ...

Journal: :Human molecular genetics 2012
Robert A Maue Robert W Burgess Bing Wang Christine M Wooley Kevin L Seburn Marie T Vanier Maximillian A Rogers Catherine C Chang Ta-Yuan Chang Brent T Harris David J Graber Carlos A A Penatti Donna M Porter Benjamin S Szwergold Leslie P Henderson John W Totenhagen Theodore P Trouard Ivan A Borbon Robert P Erickson

We have identified a point mutation in Npc1 that creates a novel mouse model (Npc1(nmf164)) of Niemann-Pick type C1 (NPC) disease: a single nucleotide change (A to G at cDNA bp 3163) that results in an aspartate to glycine change at position 1005 (D1005G). This change is in the cysteine-rich luminal loop of the NPC1 protein and is highly similar to commonly occurring human mutations. Genetic an...

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: :Journal of lipid research 2009
Jiang Wang Bei-Bei Chu Liang Ge Bo-Liang Li Yan Yan Bao-Liang Song

The Niemann-Pick C1 Like 1 (NPC1L1) is a predicted polytopic membrane protein that is critical for cholesterol absorption. NPC1L1 takes up free cholesterol into cells through vesicular endocytosis. Ezetimibe, a clinically used cholesterol absorption inhibitor, blocks the endocytosis of NPC1L1 thereby inhibiting cholesterol uptake. Human NPC1L1 is a 1,332-amino acid protein with a putative stero...

Journal: :The Journal of biological chemistry 2007
Ming-Dong Wang Vivian Franklin Meenakshi Sundaram Robert S Kiss Kenneth Ho Michel Gallant Yves L Marcel

Niemann-Pick type C1 (Npc1) protein inactivation results in lipid accumulation in late endosomes and lysosomes, leading to a defect of ATP binding cassette protein A1 (Abca1)-mediated lipid efflux to apolipoprotein A-I (apoA-I) in macrophages and fibroblasts. However, the role of Npc1 in Abca1-mediated lipid efflux to apoA-I in hepatocytes, the major cells contributing to HDL formation, is stil...

2011
Marine Hovakimyan Jana Petersen Fabian Maass Maria Reichard Martin Witt Jan Lukas Oliver Stachs Rudolf Guthoff Arndt Rolfs Andreas Wree

BACKGROUND Niemann Pick disease type C1 is a neurodegenerative disease caused by mutations in the NPC1 gene, which result in accumulation of unesterified cholesterol and glycosphingolipids in the endosomal-lysosomal system as well as limiting membranes. We have previously shown the corneal involvement in NPC1 pathology in form of intracellular inclusions in epithelial cells and keratocytes. The...

Journal: :International journal of molecular sciences 2018
Lynn Ebner Anne Gläser Anja Bräuer Martin Witt Andreas Wree Arndt Rolfs Marcus Frank Brigitte Vollmar Angela Kuhla

Niemann-Pick-disease type C1 (NPC1) is an autosomal-recessive cholesterol-storage disorder. Besides other symptoms, NPC1 patients develop liver dysfunction and hepatosplenomegaly. The mechanisms of hepatomegaly and alterations of lipid metabolism-related genes in NPC1 disease are still poorly understood. Here, we used an NPC1 mouse model to study an additive hepatoprotective effect of a combina...

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