نتایج جستجو برای: pompe disease

تعداد نتایج: 1490290  

2016
Carlos R. Ferreira William A. Gahl

Lysosomes are cytoplasmic organelles that contain a variety of different hydrolases. A genetic deficiency in the enzymatic activity of one of these hydrolases will lead to the accumulation of the material meant for lysosomal degradation. Examples include glycogen in the case of Pompe disease, glycosaminoglycans in the case of the mucopolysaccharidoses, glycoproteins in the cases of the oligosac...

Journal: :Journal of Inherited Metabolic Disease 2011

Journal: :The Journal of Pediatrics 2016

2012
Nadine AME van der Beek Juna M de Vries Marloes LC Hagemans Wim CJ Hop Marian A Kroos John HJ Wokke Marianne de Visser Baziel GM van Engelen Jan BM Kuks Anneke J van der Kooi Nicolette C Notermans Karin G Faber Jan JGM Verschuuren Arnold JJ Reuser Ans T van der Ploeg Pieter A van Doorn

BACKGROUND Due partly to physicians' unawareness, many adults with Pompe disease are diagnosed with great delay. Besides, it is not well known which factors influence the rate of disease progression, and thus disease outcome. We delineated the specific clinical features of Pompe disease in adults, and mapped out the distribution and severity of muscle weakness, and the sequence of involvement o...

2013
M Hilz U Hoppe S Moeller J Köhn

Introduction Pompe disease primarily affects skeletal and cardiac muscles. It is difficult to assess therapeutic efficacy of enzyme replacement therapy (ERT) using primary end points based on changes in weakness of large limb-girdle muscles, gait-endurance, and respiratory function, which all depend on variable day-to-day patient performance. In contrast, muscle improvement might be shown by as...

Journal: :Journal of neuromuscular diseases 2015
A Hancu N Butoianu R Rosioru

Journal: :Journal of visualized experiments : JoVE 2017
Jan Lukas Anne-Marie Knospe Susanne Seemann Valentina Citro Maria V Cubellis Arndt Rolfs

The use of personalized medicine to treat rare monogenic diseases like lysosomal storage disorders (LSDs) is challenged by complex clinical trial designs, high costs, and low patient numbers. Hundreds of mutant alleles are implicated in most of the LSDs. The diseases are typically classified into 2 to 3 different clinical types according to severity. Moreover, molecular characterization of the ...

Journal: :Molecular genetics and metabolism 2010
Karen M Ashe Kristin M Taylor Qiuming Chu Elizabeth Meyers Allen Ellis Varvara Jingozyan Katherine Klinger Patrick F Finn Christopher G F Cooper Wei-Lien Chuang John Marshall John M McPherson Robert J Mattaliano Seng H Cheng Ronald K Scheule Rodney J Moreland

Pompe disease, also known as glycogen storage disease (GSD) type II, is caused by deficiency of lysosomal acid alpha-glucosidase (GAA). The resulting glycogen accumulation causes a spectrum of disease severity ranging from a rapidly progressive course that is typically fatal by 1-2years of age to a more slowly progressive course that causes significant morbidity and early mortality in children ...

2017
Celine Baligand Adrian G. Todd Brittany Lee-McMullen Ravneet S. Vohra Barry J. Byrne Darin J. Falk Glenn A. Walter

The development of therapeutic clinical trials for glycogen storage disorders, including Pompe disease, has called for non-invasive and objective biomarkers. Glycogen accumulation can be measured in vivo with 13C MRS. However, clinical implementation remains challenging due to low signal-to-noise. On the other hand, the buildup of glycolytic intermediates may be detected with 31P MRS. We sought...

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