نتایج جستجو برای: pick type c1 npc1

تعداد نتایج: 1363808  

Journal: :Biochimica et biophysica acta 2011
Kyle B Peake Robert B Campenot Dennis E Vance Jean E Vance

Niemann-Pick Type C (NPC) disease is an autosomal recessive disorder that results in accumulation of cholesterol and other lipids in late endosomes/lysosomes and leads to progressive neurodegeneration and premature death. The mechanism by which lipid accumulation causes neurodegeneration remains unclear. Inappropriate activation of microglia, the resident immune cells of the central nervous sys...

Journal: :Journal of neuroscience research 2005
Iram Ahmad Silvia Lope-Piedrafita Xiaoning Bi Chad Hicks Yueqin Yao Clara Yu Elizabeth Chaitkin Christine M Howison Lyndon Weberg Theodore P Trouard Robert P Erickson

Niemann-Pick C disease (NPC) is an irreversible neurodegenerative disorder without current treatment. It is thought to result from deficient intracellular cholesterol and/or ganglioside trafficking. We have investigated the effects of allopregnanolone treatments on survival, weight loss, motor function, magnetic resonance imaging (MRI), and neuropathology in the mouse model of NPC (Npc1(-/-) mi...

Journal: :PLoS ONE 2006
Jonathan V. Reddy Ian G. Ganley Suzanne R. Pfeffer

BACKGROUND Niemann-Pick Type C (NPC) disease is a neurodegenerative disease that is characterized by the accumulation of cholesterol and glycosphingolipids in the late endocytic pathway. The majority of NPC cases are due to mutations in the NPC1 gene. The precise function of this gene is not yet known. METHODOLOGY/PRINCIPAL FINDINGS Using cDNA microarrays, we analyzed the genome-wide expressi...

2013
Pablo J. Sáez Juan A. Orellana Natalia Vega-Riveros Vania A. Figueroa Diego E. Hernández Juan F. Castro Andrés D. Klein Jean X. Jiang Silvana Zanlungo Juan C. Sáez

Reduced astrocytic gap junctional communication and enhanced hemichannel activity were recently shown to increase astroglial and neuronal vulnerability to neuroinflammation. Moreover, increasing evidence suggests that neuroinflammation plays a pivotal role in the development of Niemann-Pick type C (NPC) disease, an autosomal lethal neurodegenerative disorder that is mainly caused by mutations i...

2013
Mahua Maulik Gopal Thinakaran Satyabrata Kar

Niemann-Pick type C (NPC) disease, a rare autosomal recessive disorder caused mostly by mutation in NPC1 gene, is pathologically characterized by the accumulation of free cholesterol in brain and other tissues. This is accompanied by gliosis and loss of neurons in selected brain regions, including the cerebellum. Recent studies have shown that NPC disease exhibits intriguing parallels with Alzh...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2015
Jian Li Maika S Deffieu Peter L Lee Piyali Saha Suzanne R Pfeffer

Lysosomes are lined with a glycocalyx that protects the limiting membrane from the action of degradative enzymes. We tested the hypothesis that Niemann-Pick type C 1 (NPC1) protein aids the transfer of low density lipoprotein-derived cholesterol across this glycocalyx. A prediction of this model is that cells will be less dependent upon NPC1 if their glycocalyx is decreased in density. Lysosome...

Journal: :Journal of lipid research 2013
Martin Fan Rohini Sidhu Hideji Fujiwara Brett Tortelli Jessie Zhang Cristin Davidson Steven U Walkley Jessica H Bagel Charles Vite Nicole M Yanjanin Forbes D Porter Jean E Schaffer Daniel S Ory

Niemann-Pick type C (NPC)1 is a rare neurodegenerative disease for which treatment options are limited. A major barrier to development of effective treatments has been the lack of validated biomarkers to monitor disease progression or serve as outcome measures in clinical trials. Using targeted metabolomics to exploit the complex lipid storage phenotype that is the hallmark of NPC1 disease, we ...

Journal: :Human molecular genetics 2012
Matthew J Elrick Ting Yu Chan Chung Andrew P Lieberman

Niemann-Pick type C disease (NPC) is a childhood onset neurodegenerative disorder arising from lipid-trafficking defects caused by mutations in the NPC1 or NPC2 gene. Marked accumulation of autophagosomes is a prominent feature of NPC cells, yet a detailed understanding of the disease-associated alterations in autophagy and their role in pathogenesis has been lacking. Prior studies have shown t...

Journal: :PloS one 2016
Elena-Raluca Nicoli Nada Al Eisa Celine V M Cluzeau Christopher A Wassif James Gray Kathryn R Burkert David A Smith Lauren Morris Stephanie M Cologna Cody J Peer Tristan M Sissung Constantin-Daniel Uscatu William D Figg William J Pavan Charles H Vite Forbes D Porter Frances M Platt

Niemann-Pick type C (NPC) disease is a neurodegenerative lysosomal storage disease caused by mutations in either the NPC1 or NPC2 gene. NPC is characterised by storage of multiple lipids in the late endosomal/lysosomal compartment, resulting in cellular and organ system dysfunction. The underlying molecular mechanisms that lead to the range of clinical presentations in NPC are not fully underst...

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