نتایج جستجو برای: lysosomal storage disease

تعداد نتایج: 1671181  

Journal: :Indian pediatrics 2012
Prashant K Verma Prajnya Ranganath Ashwin B Dalal Shubha R Phadke

BACKGROUND There is limited literature available on the phenotypic and mutation spectrum of Indian patients with Lysosomal storage disorders (LSD). OBJECTIVE To elucidate the clinical, biochemical and mutation spectrum and to study the management options in Indian patients with lysosomal storage disorders. DESIGN Descriptive study. SUBJECTS AND METHODS All patients with lysosomal storage ...

2014
Mehwish Farrukh Ayesha Haque

Mucopolysaccharidosis (MPS) are a group of metabolic disorders of the lysosomal storage disease family caused by the absence or malfunctioning of lysosomal enzymes, which blocks degradation of mucopolysaccharides and leads to abnormal accumulation of heparan sulfate, dermatan sulfate, and keratan sulfate. Morquio’s syndrome is a rare autosomal-recessive mucopolysaccharidosis. This syndrome is c...

Journal: :Biological chemistry 2013
Na-Young Yang Yu-Na Lee He-Jin Lee Yoon Suk Kim Seung-Jae Lee

Mutations in the gene encoding glucocerebrosidase (GBA1) cause Gaucher disease (GD), a lysosomal storage disease with recessive inheritance. Glucocerebrosidase (GCase) is a lysosomal lipid hydrolase that digests glycolipid substrates, such as glucosylceramide and glucosylsphingosine. GBA1 mutations have been implicated in Lewy body diseases (LBDs), such as Parkinson's disease and dementia with ...

Journal: :Pediatrics 2009
Orna Staretz-Chacham Tess C Lang Mary E LaMarca Donna Krasnewich Ellen Sidransky

Lysosomal storage disorders are rare inborn errors of metabolism, with a combined incidence of 1 in 1500 to 7000 live births. These relatively rare disorders are seldom considered when evaluating a sick newborn. A significant number of the >50 different lysosomal storage disorders, however, do manifest in the neonatal period and should be part of the differential diagnosis of several perinatal ...

2013
Yoon Suk Kim Seung-Jae Lee

Mutations in the gene encoding glucocerebrosidase ( GBA1 ) cause Gaucher disease (GD), a lysosomal storage disease with recessive inheritance. Glucocerebrosidase (GCase) is a lysosomal lipid hydrolase that digests glycolipid substrates, such as glucosylceramide and glucosylsphingosine. GBA1 mutations have been implicated in Lewy body diseases (LBDs), such as Parkinson ’ s disease and dementia w...

Journal: :The Yale Journal of Biology and Medicine 1981
Kathleen Anne Nolan

This book is a compilation of an international symposium held in South Carolina in March 1979. It reviews the progress made during the past two decades concerning attempts at therapy of the genetic disorders which result in abnormal lysosomal enzyme metabolism. Despite the narrowness of the subject the variety of information is quite large. The first section is devoted to a discussion of enzyme...

2017
Michela Palmieri Rituraj Pal Hemanth R. Nelvagal Parisa Lotfi Gary R. Stinnett Michelle L. Seymour Arindam Chaudhury Lakshya Bajaj Vitaliy V. Bondar Laura Bremner Usama Saleem Dennis Y. Tse Deepthi Sanagasetti Samuel M. Wu Joel R. Neilson Fred A. Pereira Robia G. Pautler George G. Rodney Jonathan D. Cooper Marco Sardiello

Neurodegenerative diseases characterized by aberrant accumulation of undigested cellular components represent unmet medical conditions for which the identification of actionable targets is urgently needed. Here we identify a pharmacologically actionable pathway that controls cellular clearance via Akt modulation of transcription factor EB (TFEB), a master regulator of lysosomal pathways. We sho...

2015
Eun Jin Woo Won-Joon Lee Kyung-Seok Hu Jae Joon Hwang Clark Spencer Larsen

Skeletal dysplasias related to genetic etiologies have rarely been reported for past populations. This report presents the skeletal characteristics of an individual with dwarfism-related skeletal dysplasia from South Korea. To assess abnormal deformities, morphological features, metric data, and computed tomography scans are analyzed. Differential diagnoses include achondroplasia or hypochondro...

Journal: :Neurobiology of disease 2004
Megan Griffey Ellen Bible Carole Vogler Beth Levy Praveena Gupta Jonathan Cooper Mark S Sands

Infantile neuronal ceroid lipofuscinosis (INCL) is the earliest onset form of a class of inherited neurodegenerative disease called Batten disease. INCL is caused by a deficiency in the lysosomal enzyme palmitoyl protein thioesterase-1 (PPT1). Autofluorescent storage material accumulates in virtually all tissues in INCL patients, including the brain, and leads to widespread neuronal loss and co...

Journal: :Journal of the American Society of Nephrology : JASN 2002
Gregory M Pastores Yeong-Hau H Lien

Anderson-Fabry disease (AFD) is an inborn error of glycosphingolipid metabolism that is due to a deficiency of the lysosomal hydrolase -galactosidase A ( -Gal A) (1). It is an X-linked lysosomal storage disorder (LSD) associated with multisystemic involvement resulting from the accumulation of neutral glycosphingolipids (mostly ceramide trihexoside [CTH]) in various organs (2). The disease mani...

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