نتایج جستجو برای: pompe disease

تعداد نتایج: 1490290  

2013
Tracey Willis Mark Roberts David Hilton-Jones Ros Quinlivan Mike Hanna Volker Straub

Introduction This 12-month audit (conducted between 1 July 2011 to 30 June 2012) was designed following a retrospective audit in a specialist neuromuscular centre (UK) using DBS testing to identify late-onset Pompe disease. The objective was to screen all patients with an unknown diagnosis presenting with a limb girdle pattern of muscle weakness +/respiratory symptoms +/scoliosis +/rigid spine ...

2011
Deniz Güngör Juna M de Vries Wim CJ Hop Arnold JJ Reuser Pieter A van Doorn Ans T van der Ploeg Marloes LC Hagemans

BACKGROUND Pompe disease is a rare lysosomal storage disorder characterized by muscle weakness and wasting. The majority of adult patients have slowly progressive disease, which gradually impairs mobility and respiratory function and may lead to wheelchair and ventilator dependency. It is as yet unknown to what extent the disease reduces the life span of these patients. Our objective was to det...

Journal: :Journal of Neuromuscular Diseases 2015

Journal: :Revista portuguesa de pneumologia 2017
M J Guimarães J C Winck B Conde A Mineiro M Raposo J Moita A Marinho J M Silva N Pires S André C Loureiro

Pompe disease is a rare autosomal recessive neuromuscular disorder caused by acid α-glucosidase enzyme (GAA) deficiency and divided into two distinct variants, infantile- and late-onset. The late-onset variant is characterized by a spectrum of phenotypic variation that may range from asymptomatic, to reduced muscle strength and/or diaphragmatic paralysis. Since muscle strength loss is character...

2012
George Papadimas Gerassimos Terzis Constantinos Papadopoulos Anna Areovimata Konstantinos Spengos Stavros Kavouras Panagiota Manta

BACKGROUND Pompe disease is an inherited metabolic disorder characterized by α-glycosidase deficiency, which leads to lysosomal glycogen accumulation in many different tissues. The infantile form is the most severe with a rapidly fatal outcome, while the late onset form has a greater phenotypic variability, characterized by skeletal muscle dysfunction and early respiratory involvement. Bone min...

Journal: :Circulation. Cardiovascular genetics 2016
Stephan C A Wens Gerben J Schaaf Michelle Michels Michelle E Kruijshaar Tom J M van Gestel Stijn In 't Groen Joon Pijnenburg Dick H W Dekkers Jeroen A A Demmers Lex B Verdijk Esther Brusse Ron H N van Schaik Ans T van der Ploeg Pieter A van Doorn W W M Pim Pijnappel

BACKGROUND Elevated plasma cardiac troponin T (cTnT) levels in patients with neuromuscular disorders may erroneously lead to the diagnosis of acute myocardial infarction or myocardial injury. METHODS AND RESULTS In 122 patients with Pompe disease, the relationship between cTnT, cardiac troponin I, creatine kinase (CK), CK-myocardial band levels, and skeletal muscle damage was assessed. ECG an...

Journal: :Journal of neuromuscular diseases 2015
Thomas Hundsberger Kai M Rösler Oliver Findling

Enzyme replacement therapy (ERT) with recombinant human alglucosidase alfa (rhGAA) in Pompe disease is moderately effective and a life-long therapy is warranted. Clinical investigations of temporary ERT interruption are lacking, but might be of clinical signifi cance (i.e. due to patient’s wish, adherence issues, holidays, or problems with drug supply). In Switzerland, ERT for Pompe disease was...

Journal: :The Biochemical journal 2005
Yunxiang Zhu Xuemei Li Alison McVie-Wylie Canwen Jiang Beth L Thurberg Nina Raben Robert J Mattaliano Seng H Cheng

To enhance the delivery of rhGAA (recombinant GAA, where GAA stands for acid alpha-glucosidase) to the affected muscles in Pompe disease, the carbohydrate moieties on the enzyme were remodelled to exhibit a high affinity ligand for the CI-MPR (cation-independent M6P receptor, where M6P stands for mannose 6-phosphate). This was achieved by chemically conjugating on to rhGAA, a synthetic oligosac...

Journal: :Journal of neuromuscular diseases 2015
Thaís Rodrigues Carlo D Marrone

Pompe disease (glycogen storage disease type II, glycogenosis II, or acid maltase defi ciency) is a lysosomal storage disorder in which an alpha-glucosidase (GAA) defi ciency causes intralysosomal accumulation of glycogen in all tissues, notably skeletal muscles. Pompe disease is transmitted as an autosomal recessive trait and is caused by mutations in the gene encoding the GAA, located on chro...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید