نتایج جستجو برای: lysosomal storage disease

تعداد نتایج: 1671181  

2017
Daniel K Borger Benjamin McMahon Tamanna Roshan Lal Jenny Serra-Vinardell Elma Aflaki Ellen Sidransky

Induced pluripotent stem cells (iPSCs) have provided new opportunities to explore the cell biology and pathophysiology of human diseases, and the lysosomal storage disorder research community has been quick to adopt this technology. Patient-derived iPSC models have been generated for a number of lysosomal storage disorders, including Gaucher disease, Pompe disease, Fabry disease, metachromatic ...

2016
Barbara Canonico Erica Cesarini Sara Salucci Francesca Luchetti Elisabetta Falcieri Gianna Di Sario Fulvio Palma Stefano Papa

Niemann-Pick disease type A (NP-A) and type B (NP-B) are lysosomal storage diseases (LSDs) caused by sphingomyelin accumulation in lysosomes relying on reduced or absent acid sphingomyelinase. A considerable body of evidence suggests that lysosomal storage in many LSD impairs autophagy, resulting in the accumulation of poly-ubiquitinated proteins and dysfunctional mitochondria, ultimately leadi...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2004
Yun-Ping Wu Richard L Proia

Sandhoff disease is a prototypical lysosomal storage disorder in which a heritable deficiency of a lysosomal enzyme, beta-hexosaminidase, results in the storage of the enzyme's substrates in lysosomes. As with many of the other lysosomal storage diseases, neurodegeneration is a prominent feature. Although the cellular and molecular pathways that underlie the neurodegenerative process are not ye...

Journal: :Trends in Neurosciences 2011

Journal: :Muscle & nerve. Supplement 1995
A J Reuser M A Kroos M M Hermans A G Bijvoet M P Verbeet O P Van Diggelen W J Kleijer A T Van der Ploeg

Glycogen storage disease type II (GSD II/glycogenosis type II/Pompe's disease/acid maltase deficiency) is caused by the deficiency of lysosomal alpha-glucosidase resulting in lysosomal accumulation of glycogen. The disease is inherited as an autosomal recessive trait and is clinically heterogeneous. Early and late onset phenotypes are distinguished. Insight in the molecular nature of the lysoso...

Journal: :JIMD reports 2014
Jayesh Sheth Mehul Mistri Frenny Sheth Raju Shah Ashish Bavdekar Koumudi Godbole Nidhish Nanavaty Chaitanya Datar Mahesh Kamate Nrupesh Oza Chitra Ankleshwaria Sanjeev Mehta Marie Jackson

Lysosomal storage disorders (LSDs) are considered to be a rare metabolic disease for the national health forum, clinicians, and scientists. This study aimed to know the prevalence of different LSDs, their geographical variation, and burden on the society. It included 1,110 children from January 2002 to December 2012, having coarse facial features, hepatomegaly or hepatosplenomegaly, skeletal dy...

Journal: :Cold Spring Harbor Perspectives in Biology 2011

Journal: :Journal of clinical pathology 2001
W van der Meer B S Jakobs G Bocca J A Smeitink J H Schuurmans Steckhoven M H de Keijzer

In general, peripheral blood smears are performed to obtain information with regard to various morphological features as an aid in the diagnosis of infection or malignancy. This report presents a patient with I cell disease (inclusion cell disease), a fatal lysosomal storage disorder caused by a defect in an enzyme responsible for the transfer of mannose-6-phosphate ligands to precursor lysosom...

2013
R Violano M Ripolone V Lucchini L Villa M Sciacco G Comi P Tonin M Filosto S Previtali T Mongini L Vercelli E Vittonatto A Toscano O Musumeci E Barca C Angelini S Ravaglia C Lamperti M Mora L Morandi M Moggio

Introduction Glycogen storage disease, glycogenosis type II (GSDII), or Pompe disease (OMIM 23230), is an autosomal recessive lysosomal storage disorder that results from a deficiency in the acid alpha glucosidase (GAA) enzyme. The disease is characterized by progressive accumulation of lysosomal glycogen in various tissues, primarily in cardiac and skeletal muscles. The histopathological hallm...

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