نتایج جستجو برای: lysosomal storage disease

تعداد نتایج: 1671181  

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...

2016
Jinlong Jian Qing-Yun Tian Aubryanna Hettinghouse Shuai Zhao Helen Liu Jianlu Wei Gabriele Grunig Wujuan Zhang Kenneth D.R. Setchell Ying Sun Herman S. Overkleeft Gerald L. Chan Chuan-ju Liu

Gaucher disease (GD), the most common lysosomal storage disease, is caused by mutations in GBA1 encoding of β-glucocerebrosidase (GCase). Recently it was reported that progranulin (PGRN) insufficiency and deficiency associated with GD in human and mice, respectively. However the underlying mechanisms remain unknown. Here we report that PGRN binds directly to GCase and its deficiency results in ...

Journal: :Biochimica et Biophysica Acta (BBA) - Molecular Basis of Disease 1995

Journal: :British Journal of Haematology 2005

Journal: :Neurology 2011
Edward M Kaye

319 Olfaction in Parkin heterozygotes and compound heterozygotes: The CORE-PD study R.N. Alcalay, A. Siderowf, R. Ottman, E. Caccappolo, H. Mejia-Santana, M.-X. Tang, L. Rosado, E. Louis, D. Ruiz, C. Waters, S. Fahn, L. Cote, S. Frucht, B. Ford, M. Orbe-Reilly, B. Ross, M. Verbitsky, S. Kisselev, C. Comella, A. Colcher, D. Jennings, M. Nance, S. Bressman, W.K. Scott, C. Tanner, S. Mickel, M. Re...

Journal: :European journal of internal medicine 2013
Daniel Franzen Pierre A Krayenbuehl Olivier Lidove John-David Aubert Frederic Barbey

Fabry disease (FD) is an X-linked lysosomal storage disorder caused by deficiency of alpha-galactosidase A, which leads to storage of sphingolipids in virtually all human cells and consequently to organ dysfunction. Pulmonary involvement is still debated. But, obstructive lung disease is up to ten times more prevalent in patients with FD compared to general public. Also, an accelerated decline ...

2016
Heike Wolf Markus Damme Stijn Stroobants Rudi D'Hooge Hans Christian Beck Irm Hermans-Borgmeyer Renate Lüllmann-Rauch Thomas Dierks Torben Lübke

Fucosidosis is a rare lysosomal storage disorder caused by the inherited deficiency of the lysosomal hydrolase α-L-fucosidase, which leads to an impaired degradation of fucosylated glycoconjugates. Here, we report the generation of a fucosidosis mouse model, in which the gene for lysosomal α-L-fucosidase (Fuca1) was disrupted by gene targeting. Homozygous knockout mice completely lack α-L-fucos...

2009
Maryam Banikazemi

of various cells, such as in the vascular endothelium of multiple organs (1). The accumulation of GL-3 in the lysosomes causes lysosomal and cellular dysfunction and this in turn, triggers the cascade of cellular and tissue ischemia and fibrosis. The estimated prevalence of Fabry disease is about one in every 117000 live born males. The classic phenotype of Fabry disease is seen Introduction Fa...

Journal: :FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2005
Gary Hin-Fai Yam Christian Zuber Jürgen Roth

Mutations in proteins that induce misfolding and proteasomal degradation are common causes of inherited diseases. Fabry disease is a lysosomal storage disorder caused by a deficiency of alpha-galactosidase A activity in lysosomes resulting in an accumulation of glycosphingolipid globotriosylceramide (Gb3). Some classical Fabry hemizygotes and all cardiac variants have residual alpha-galactosida...

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