نتایج جستجو برای: lysosomal disorders

تعداد نتایج: 684439  

Journal: :Trends in biotechnology 2002
Aubrey D N J de Grey

Lysosomal degradation of damaged macromolecules is imperfect: many cell types accumulate lysosomal aggregates with age. Some such deposits are known, or are strongly suspected, to cause age-related disorders such as atherosclerosis and neurodegeration. It is possible that they also influence the rate of aging in general. Lysosomal degradation involves extensive cooperation between the participa...

1997
Thomas BERG Ole K. TOLLERSRUD Steven U. WALKLEY Donald SIEGEL Øivind NILSSEN

α-Mannosidosis is a lysosomal storage disorder that is caused by the deficiency of lysosomal α-mannosidase. Feline αmannosidosis is a well-characterized animal model used for studying pathological and therapeutic aspects of lysosomal storage disorders. We here report the purification of feline liver lysosomal α-mannosidase and determination of its cDNA sequence. The active enzyme consisted of t...

2013
Carmine Settembre Andrea Ballabio

Lysosomal storage disorders (LSDs) are inherited diseases characterized by progressive intracellular accumulation of undigested macromolecules within the cell due to specific lysosomal defects. Lysosomal storage results in a global impairment ofmany lysosome‐dependent pathways (e.g. autophagy and endocytosis), leading to cellular dysfunction and death (Ballabio &Gieselmann, 2009). LSD patients ...

2012
Gheona Altarescu Rachel Beeri Rachel Eiges Silvina Epsztejn-Litman Talia Eldar-Geva Deborah Elstein Ari Zimran Ehud J. Margalioth Ephrat Levy-Lahad Paul Renbaum

Preimplantation genetic diagnosis (PGD) allows birth of unaffected children for couples at risk for a genetic disorder. We present the strategy and outcome of PGD for four lysosomal storage disorders (LSD): Tay-Sachs disease (TSD), Gaucher disease (GD), Fabry disease (FD), and Hunter syndrome (HS), and subsequent development of stem cell lines. For each disease, we developed a family-specific f...

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...

2017
Sabrina Congedi Chiara Di Pede Maurizio Scarpa Angelica Rampazzo Franca Benini

Mucopolysaccharidoses (MPSs) are a group of rare, genetic lysosomal storage disorders. They are caused by deficiencies of the lysosomal enzymes involved in the degradation of glycosaminoglycans (GAGs). Pain is a common feature in mucopolysaccharidoses. However, the pathophysiology of pain in this group of diseases is still unclear and genesis of pain is multifactorial. Currently, poor data abou...

2017
Zahid Padamsey Lindsay McGuinness Nigel J. Emptage

A growing body of evidence suggests that lysosomes, which have traditionally been regarded as degradative organelles, can function as Ca2+ stores, regulated by the second messenger nicotinic acid adenine dinucleotide phosphate (NAADP). We previously demonstrated that in hippocampal pyramidal neurons, activity-dependent Ca2+ release from these stores triggers fusion of the lysosome with the plas...

2017
Daniel K Borger Benjamin McMahon Tamanna Roshan Lal Jenny Serra-Vinardell Elma Aflaki Ellen Sidransky

Induced pluripotent stem cells (iPSCs) have provided new opportunities to explore the cell biology and pathophysiology of human diseases, and the lysosomal storage disorder research community has been quick to adopt this technology. Patient-derived iPSC models have been generated for a number of lysosomal storage disorders, including Gaucher disease, Pompe disease, Fabry disease, metachromatic ...

Journal: :JIMD reports 2014
Jayesh Sheth Mehul Mistri Frenny Sheth Raju Shah Ashish Bavdekar Koumudi Godbole Nidhish Nanavaty Chaitanya Datar Mahesh Kamate Nrupesh Oza Chitra Ankleshwaria Sanjeev Mehta Marie Jackson

Lysosomal storage disorders (LSDs) are considered to be a rare metabolic disease for the national health forum, clinicians, and scientists. This study aimed to know the prevalence of different LSDs, their geographical variation, and burden on the society. It included 1,110 children from January 2002 to December 2012, having coarse facial features, hepatomegaly or hepatosplenomegaly, skeletal dy...

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