نتایج جستجو برای: infantile pompe disease

تعداد نتایج: 1498901  

2011
Simona Fecarotta Giancarlo Parenti Serena Ascione Giuseppe Montefusco Generoso Andria

Pompe disease (PD) is a rare metabolic myopathy. It is caused by the deficiency of the lysosomal enzyme acid alpha glucosidase (GAA), with a consequent generalized storage of glycogen, particularly in the heart, skeletal muscle and liver. It has been reported an overall incidence of 1 in 40.000 live birth, with a different frequency in different races. The infantile form has an incidence of 1 i...

Journal: :Circulation. Cardiovascular imaging 2015
Sushil P Tripathi Milind S Phadke Prafulla G Kerkar

A 5-month-old male infant born to consanguinious marriage with past history of feeding difficulties in the form of suck-rest-suck cycle, forehead diaphoresis, and poor weight gain since early infancy presented with increasing severity of respiratory distress and apathy of 2 weeks duration. Physical examination revealed tachypnea, tachycardia, severe respiratory distress with chest wall retracti...

Journal: :Molecular therapy : the journal of the American Society of Gene Therapy 2010
Baodong Sun Michael D Kulis Sarah P Young Amy C Hobeika Songtao Li Andrew Bird Haoyue Zhang Yifan Li Timothy M Clay Wesley Burks Priya S Kishnani Dwight D Koeberl

Infantile Pompe disease progresses to a lethal cardiomyopathy in absence of effective treatment. Enzyme-replacement therapy (ERT) with recombinant human acid alpha-glucosidase (rhGAA) has been effective in most patients with Pompe disease, but efficacy was reduced by high-titer antibody responses. Immunomodulatory gene therapy with a low dose adeno-associated virus (AAV) vector (2 x 10(10) part...

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: :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...

2015
Lisa D. Hobson-Webb Stephanie L. Austin Sneha Jain Laura E. Case Karla Greene Priya S. Kishnani

BACKGROUND Prior autopsy reports demonstrate glycogen deposition in Schwann cells of the peripheral nerves in patients with infantile and late-onset Pompe disease (LOPD), but little is known about associated clinical features. CASE REPORT Here, we report the first confirmed cases of small-fiber neuropathy (SFN) in LOPD and present the results of a first attempt at screening for SFN in this pa...

2013
I Tournev T Chamova I Sinigerska V Guergueltcheva M Gospodinova P Ganova R Stoilov A Todorova T Todorov

Introduction Pompe disease is a rare, metabolic, multi-system, lysosomal storage disorder with autosomal recessive inheritance, caused by a deficiency of the glycogen-degrading lysosomal enzyme, acid alpha-glucosidase (GAA). Great phenotypic variability has led to the classification of several subtypes: infantile, late-infantile, childhood, juvenile, and adult-onset form, based on the age of on...

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