نتایج جستجو برای: lysosomal myopathy

تعداد نتایج: 27249  

2011
Adam Jara

Pompe disease is an inherited, autosomal-recessive deficiency of acid alpha-glucosidase (GAA), a lysosomal enzyme required for the breakdown of glycogen [1]. The prevailing pathology is a generalized build-up of glycogen within lysosomes leading to profound skeletal and cardiac myopathy with the severity of symptoms being directly correlated with residual GAA activity [1]. In the most severe fo...

Journal: :The New England journal of medicine 2008
E Link S Parish J Armitage L Bowman S Heath F Matsuda I Gut M Lathrop R Collins

BACKGROUND Lowering low-density lipoprotein cholesterol with statin therapy results in substantial reductions in cardiovascular events, and larger reductions in cholesterol may produce larger benefits. In rare cases, myopathy occurs in association with statin therapy, especially when the statins are administered at higher doses and with certain other medications. METHODS We carried out a geno...

Journal: :American heart journal 2010
Soko Setoguchi John M Higgins Helen Mogun Vamsi K Mootha Jerry Avorn

BACKGROUND A recent large-scale, chemical screening study raised the hypothesis that propranolol may increase the risk of myopathy. We tested this hypothesis in a large population to assess whether (1) propranolol use is associated with an increased risk of myopathy and (2) the concurrent use of propranolol with a statin may further increase risk of myopathy. METHODS New users of propranolol ...

2010
Shoichi Takikita Cynthia Schreiner Rebecca Baum Tao Xie Evelyn Ralston Paul H. Plotz Nina Raben

PGC-1α is a transcriptional co-activator that plays a central role in the regulation of energy metabolism. Our interest in this protein was driven by its ability to promote muscle remodeling. Conversion from fast glycolytic to slow oxidative fibers seemed a promising therapeutic approach in Pompe disease, a severe myopathy caused by deficiency of the lysosomal enzyme acid alpha-glucosidase (GAA...

Journal: :Medicine 2005
Barbara C Sonies Phaedra Almajid Robert Kleta Isa Bernardini William A Gahl

Nephropathic cystinosis is a rare, autosomal recessive lysosomal storage disorder caused by mutations in the CTNS gene that codes for a cystine transporter in the lysosomal membrane. Affected patients store 50-100 times the normal amounts of cystine in their cells, and suffer renal tubular and glomerular disease, growth retardation, photophobia, and other systemic complications, including a myo...

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....

Journal: :Proceedings of the Royal Society of Medicine 1913

Journal: :Journal of Neurology, Neurosurgery & Psychiatry 1979

2018
Zhiyv Niu Carly Sabine Pontifex Sarah Berini Leslie E. Hamilton Elie Naddaf Eric Wieben Ross A. Aleff Kristina Martens Angela Gruber Andrew G. Engel Gerald Pfeffer Margherita Milone

Objective The aim of this study is to identify the molecular defect of three unrelated individuals with late-onset predominant distal myopathy; to describe the spectrum of phenotype resulting from the contributing role of two variants in genes located on two different chromosomes; and to highlight the underappreciated complex forms of genetic myopathies. Patients and methods Clinical and labo...

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