نتایج جستجو برای: pompe disease

تعداد نتایج: 1490290  

2013
Stephanie Austin Priya Kishnani

Pompe disease (glycogen storage disease type II) is an autosomal recessive lysosomal storage disorder caused by a deficiency of the enzyme acid alpha-glucosidase. Enzyme replacement therapy (ERT) with alglucosidase alfa has resulted in a clinical benefit in a subset of patients. Crossreactive Immunological Material (CRIM)-negative status is associated with poor prognosis. Patients with CRIM-neg...

2017
Derya Ceyhan Burcu Gucyetmez Topal

We aim to create an information platform by contributing orodental findings of Pompe disease to literature. An 18-month-old male patient with Pompe disease was referred to our clinic due to swelling of the gums. In first dental examination, a nonfluctuant, normal gingiva colored swelling at the right anterior region of maxilla was detected. His parents were recommended to perform finger massage...

2013
CARMEN ESMER ROSARIO BECERRA-BECERRA CLAUDIA PEÑA-ZEPEDA ANTONIO BRAVO-ORO

Glycogen-storage disease type II, also named Pompe disease, is caused by the deficiency of the enzyme acid alpha-glucosidase, which originates lysosomal glycogen accumulation leading to progressive neuromuscular damage. Early-onset Pompe disease shows a debilitating and frequently fulminating course. To date, more than 300 mutations have been described; the majority of them are unique to each a...

Journal: :Molecular genetics and metabolism 2016
Ans van der Ploeg Pierre G Carlier Robert-Yves Carlier John T Kissel Benedikt Schoser Stephan Wenninger Alan Pestronk Richard J Barohn Mazen M Dimachkie Ozlem Goker-Alpan Tahseen Mozaffar Loren D M Pena Zachary Simmons Volker Straub Michela Guglieri Peter Young Matthias Boentert Pierre-Yves Baudin Stephan Wens Raheel Shafi Carl Bjartmar Beth L Thurberg

BACKGROUND Late-onset Pompe disease is characterized by progressive skeletal myopathy followed by respiratory muscle weakness, typically leading to loss of ambulation and respiratory failure. In this population, enzyme replacement therapy (ERT) with alglucosidase alfa has been shown to stabilize respiratory function and improve mobility and muscle strength. Muscle pathology and glycogen clearan...

Journal: :Pediatrics 2009
Yin-Hsiu Chien Ni-Chung Lee Beth L Thurberg Shu-Chuan Chiang Xiaokui Kate Zhang Joan Keutzer Ai-Chu Huang Mei-Hwan Wu Pei-Hsin Huang Fuu-Jen Tsai Yuan-Tsong Chen Wuh-Liang Hwu

OBJECTIVE Pompe disease causes progressive, debilitating, and often life-threatening musculoskeletal, respiratory, and cardiac symptoms. Favorable outcomes with early intravenous enzyme-replacement therapy and alglucosidase alfa have been reported, but early clinical diagnosis before the development of severe symptoms has rarely been possible in infants. METHODS We recently conducted a newbor...

Journal: :BMC Musculoskeletal Disorders 2013

Journal: :Human Molecular Genetics 2011

Journal: :Kathmandu University medical journal 2015
S Koirala A Poudel R Basnet K Subedi

Infantile hypotonia or floppy infant is a diagnostic challenge when it presents with other presenting complaints such as fever, cough or diarrhea. Many times the hypotonia goes unnoticed when other symptom covers the hypotonia and child continues to receive the treatment for other symptoms. We report a rare case from Nepal of infantile Pompe disease who presented with the history of fever and c...

Journal: :FASEB journal : official publication of the Federation of American Societies for Experimental Biology 2014
Dwight D Koeberl Stephanie Austin Laura E Case Edward C Smith Anne F Buckley Sarah P Young Deeksha Bali Priya S Kishnani

Effective dosages for enzyme replacement therapy (ERT) in Pompe disease are much higher than for other lysosomal storage disorders, which has been attributed to low cation-independent mannose-6-phosphate receptor (CI-MPR) in skeletal muscle. We have previously demonstrated the benefit of increased CI-MPR-mediated uptake of recombinant human acid-α-glucosidase during ERT in mice with Pompe disea...

Journal: :Journal of neuromuscular diseases 2015
Mark Tarnopolsky

patients with Pompe disease, careful monitoring of patients is also essential for optimal care. Guidelines for the care of patients with Pompe disease have been published by a number of countries and the goal of the presentation is to highlight some of the consensus recommendations. The goal of guidelines is to provide a general framework of evidence or consensus-based standards of care for the...

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