نتایج جستجو برای: pompe disease

تعداد نتایج: 1490290  

Journal: :International Journal of Neonatal Screening 2020

2012
Andreas Herzog Ralf Hartung Arnold J J Reuser Pia Hermanns Heiko Runz Nesrin Karabul Seyfullah Gökce Joachim Pohlenz Christoph Kampmann Christina Lampe Michael Beck Eugen Mengel

BACKGROUND Pompe disease (Glycogen storage disease type II, GSD II, acid alpha-glucosidase deficiency, acid maltase deficiency, OMIM # 232300) is an autosomal-recessive lysosomal storage disorder due to a deficiency of acid alpha-glucosidase (GAA, acid maltase, EC 3.2.1.20, Swiss-Prot P10253). Clinical manifestations are dominated by progressive weakness of skeletal muscle throughout the clinic...

2013
N Karabul S Gökce M Kirchner W Mannhardt E Mengel

Introduction Glycogen storage disease type II (Pompe disease or acid maltase deficiency) is an autosomal recessive metabolic disorder caused by a deficiency of the lysosomal enzyme acid alpha-glucosidase. Accumulation of glycogen in the lysosomes damages muscle cells throughout the body. In response to that damage, we hypothesized that cytokines (a family of proteins that mediate innate and ada...

2014
Richie Khanna Allan C. Powe Yi Lun Rebecca Soska Jessie Feng Rohini Dhulipala Michelle Frascella Anadina Garcia Lee J. Pellegrino Su Xu Nastry Brignol Matthew J. Toth Hung V. Do David J. Lockhart Brandon A. Wustman Kenneth J. Valenzano

Pompe disease is an inherited lysosomal storage disorder that results from a deficiency in acid α-glucosidase (GAA) activity due to mutations in the GAA gene. Pompe disease is characterized by accumulation of lysosomal glycogen primarily in heart and skeletal muscles, which leads to progressive muscle weakness. We have shown previously that the small molecule pharmacological chaperone AT2220 (1...

Journal: :The Journal of biological chemistry 2004
Yunxiang Zhu Xuemei Li Josephine Kyazike Qun Zhou Beth L Thurberg Nina Raben Robert J Mattaliano Seng H Cheng

Clinical studies of enzyme replacement therapy for Pompe disease have indicated that relatively high doses of recombinant human acid alpha-glucosidase (rhGAA) may be required to reduce the abnormal glycogen storage in cardiac and skeletal muscles. This may be because of inefficient cation-independent mannose 6-phosphate receptor (CI-MPR)-mediated endocytosis of the enzyme by the affected target...

Journal: :Molecular genetics and metabolism 2010
Piers C A Barker Sara K Pasquali Stephen Darty Richard J Ing Jennifer S Li Raymond J Kim Stephanie DeArmey Priya S Kishnani Michael J Campbell

BACKGROUND Pompe disease (acid α-glucosidase deficiency) is one of several lysosomal storage diseases amenable to treatment with enzyme replacement therapy (ERT). While echocardiography (echo) has been the standard method to evaluate the cardiac response to ERT, cardiac magnetic resonance imaging (CMR) has the advantage of a better tissue definition and characterization of myocardial fibrosis. ...

2018
Yasuyuki Fukuhara Naoko Fuji Narutoshi Yamazaki Asami Hirakiyama Tetsuharu Kamioka Joo-Hyun Seo Ryuichi Mashima Motomichi Kosuga Torayuki Okuyama

Pompe disease is an autosomal recessive disorder caused by acid α-glucosidase (GAA) deficiency, which results in the accumulation of glycogen in lysosomes in multiple tissues, including cardiac, skeletal, and smooth muscle cells. Thus far, 558 sequence variants of the GAA gene have been published in the Pompe Disease Mutation Database, and some mutations appear with considerable frequency in pa...

2014
Istvan Katona Joachim Weis Frank Hanisch

BACKGROUND Glycogenosis type II or Pompe disease is an autosomal-recessive lysosomal storage disease due to mutations in the gene encoding acid alpha-glucosidase (GAA), an enzyme required for lysosomal glycogen degradation. The disease predominantly affects the skeletal and respiratory muscles but there is growing evidence of the involvement of smooth muscle cells in blood vessel walls, suggest...

Journal: :Human molecular genetics 2011
Barry J Byrne Darin J Falk Christina A Pacak Sushrusha Nayak Roland W Herzog Melissa E Elder Shelley W Collins Thomas J Conlon Nathalie Clement Brian D Cleaver Denise A Cloutier Stacy L Porvasnik Saleem Islam Mai K Elmallah Anatole Martin Barbara K Smith David D Fuller Lee Ann Lawson Cathryn S Mah

Pompe disease is an autosomal recessive metabolic myopathy caused by the deficiency of the lysosomal enzyme acid alpha-glucosidase and results in cellular lysosomal and cytoplasmic glycogen accumulation. A wide spectrum of disease exists from hypotonia and severe cardiac hypertrophy in the first few months of life due to severe mutations to a milder form with the onset of symptoms in adulthood....

2013
Stephan C A Wens Carin M van Gelder Michelle E Kruijshaar Juna M de Vries Nadine A M E van der Beek Arnold J J Reuser Pieter A van Doorn Ans T van der Ploeg Esther Brusse

BACKGROUND Pompe disease has a broad clinical spectrum, in which the phenotype is partially explained by the genotype. The aim of this study was to describe phenotypical variation among siblings with non-classic Pompe disease. We hypothesized that siblings and families with the same genotype share more similar phenotypes than the total population of non-classic Pompe patients, and that this mig...

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