نتایج جستجو برای: lysosomal storage disease

تعداد نتایج: 1671181  

Journal: :Blood 1991
Joel Charrow

Gaucher disease is the most common lysosomal storage disease, and the first lysosomal storage disease for which a specific therapy has been developed. Enzyme replacement therapy, with glucocerebrosidase purified from human placentae, was introduced in 1991. Recombinant human glucocerebrosidase, produced by Chinese hamster ovary cells in tissue culture, became available in 1994 and has replaced ...

Journal: :The Journal of biological chemistry 2012
Miao Xu Ke Liu Manju Swaroop Forbes D Porter Rohini Sidhu Sally Firnkes Daniel S Ory Juan J Marugan Jingbo Xiao Noel Southall William J Pavan Cristin Davidson Steven U Walkley Alan T Remaley Ulrich Baxa Wei Sun John C McKew Christopher P Austin Wei Zheng

Niemann-Pick disease type C (NPC) and Wolman disease are two members of a family of storage disorders caused by mutations of genes encoding lysosomal proteins. Deficiency in function of either the NPC1 or NPC2 protein in NPC disease or lysosomal acid lipase in Wolman disease results in defective cellular cholesterol trafficking. Lysosomal accumulation of cholesterol and enlarged lysosomes are s...

Journal: :Veterinary pathology 1987
H J Baker P A Wood D A Wenger S U Walkley K Inui T Kudoh M C Rattazzi B L Riddle

A 7-month-old Balinese cat with progressive neurological dysfunction had histopathological lesions of brain, liver, kidney, spleen, and lung consistent with a lysosomal storage disease. Ultrastructural examination revealed lysosomal hypertrophy with membranous inclusions. Hepatic sphingomyelin and cholesterol were elevated 10 times normal, and total phospholipids were increased 3.6 fold. Sphing...

Journal: :Journal of child neurology 1996
A Prasad E M Kaye J Alroy

In this report, we have summarized our 9-year experience of over 100 proven cases of lysosomal storage disease using electron microscopic evaluation of skin biopsies as a screening tool. The skin biopsy was very specific in establishing the diagnosis in only two disorders, namely neuronal ceroid lipofuscinosis and mucolipidosis IV. Although the biopsy was not diagnostic in other categories of s...

عمادی, عاطفه , فکری, کیاوش , نوربخش, محمد کاظم , نکویی, افسانه , هاشمی دهکردی, الهام ,

Gangliosidosis GM1, is a lipid storage disorder due to deficiency of the lysosomal enzyme &beta-galactosidase. This deficiency leads to lysosomal accumulation of gangliosidosis GM1 to be, which contributes to swelling, cell damage and dysfunction of the organs. Gangliosidosis GM1a rare neurodegenerative disorder. Mongolian spots are significant clinical sign in some of the metabolic diseases, s...

Journal: :Heart 2007
Ales Linhart Perry M Elliott

Correspondence to: Dr Perry Elliott, The Heart Hospital, 16–18 Westmoreland Street, London, W1G 8PH, UK; [email protected] __________________________ L ysosomal storage disorders (LSD) comprise a group of more than 40 diseases caused by a deficiency of lysosomal enzymes, membrane transporters or other proteins involved in lysosomal biology. The predominant inheritance pattern is autosomal...

Journal: :Physiology 2010
Emma J Parkinson-Lawrence Tetyana Shandala Mark Prodoehl Revecca Plew Glenn N Borlace Doug A Brooks

The discovery over five decades ago of the lysosome, as a degradative organelle and its dysfunction in lysosomal storage disorder patients, was both insightful and simple in concept. Here, we review some of the history and pathophysiology of lysosomal storage disorders to show how they have impacted on our knowledge of lysosomal biology. Although a significant amount of information has been acc...

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