نتایج جستجو برای: iduronidase enzyme deficiency

تعداد نتایج: 368943  

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2014
Christian Hinderer Peter Bell Brittney L Gurda Qiang Wang Jean-Pierre Louboutin Yanqing Zhu Jessica Bagel Patricia O'Donnell Tracey Sikora Therese Ruane Ping Wang Mark E Haskins James M Wilson

Patients with mucopolysaccharidosis type I (MPS I), a genetic deficiency of the lysosomal enzyme α-l-iduronidase (IDUA), exhibit accumulation of glycosaminoglycans in tissues, with resulting diverse clinical manifestations including neurological, ocular, skeletal, and cardiac disease. MPS I is currently treated with hematopoietic stem cell transplantation or weekly enzyme infusions, but these t...

2017
Natalie S Rodriguez Lisa Yanuaria Kevin Murphy R Parducho Irving M Garcia Bino A Varghese Brendan H Grubbs Toshio Miki

Mucopolysaccharidosis type 1 (MPS1) is an inherited lysosomal storage disorder caused by a deficiency in the glycosaminoglycan (GAG)-degrading enzyme α-l-iduronidase (IDUA). In affected patients, the systemic accumulation of GAGs results in skeletal dysplasia, neurological degeneration, multiple organ dysfunction, and early death. Current therapies, including enzyme replacement and bone marrow ...

2015
Yohei Sato Masako Fujiwara Hiroshi Kobayashi Michio Yoshitake Kazuhiro Hashimoto Yuji Oto Hiroyuki Ida

Mucopolysaccharidosis (MPS) is an inherited metabolic disease caused by deficiency of the enzymes needed for glycosaminoglycan (GAG) degradation. MPS type I is caused by the deficiency of the lysosomal enzyme alpha-l-iduronidase and is classified into Hurler syndrome, Scheie syndrome, and Hurler-Scheie syndrome based on disease severity and onset. Cardiac complications such as left ventricular ...

Journal: :Proceedings of the National Academy of Sciences of the United States of America 2008
Chi-hong B Chen Kenneth R Dellamaggiore Christopher P Ouellette Cecilia D Sedano Meikana Lizadjohry George A Chernis Michelle Gonzales Francis E Baltasar Audrey L Fan Rachel Myerowitz Elizabeth F Neufeld

Enzyme replacement therapy for lysosomal storage diseases is currently based on endocytosis of lysosomal enzymes via the mannose or mannose 6-phosphate receptors. We are developing a technology for endocytosis of lysosomal enzymes that depends on generic, chemically conjugated reagents. These reagents are aptamers (single-stranded nucleic acid molecules) selected to bind to the extracellular do...

Journal: :The Journal of pediatrics 2005
Jennifer Conway Sarah Dyack Bruce N A Crooks Conrad V Fernandez

Hurler syndrome is a lysosomal storage disease resulting in fatal cardiac or neurologic sequelae unless alpha-iduronidase production is reconstituted with hematopoietic stem cell transplantation. We report on a 4-year, 6-month-old boy with mixed donor chimerism and low enzyme levels but a normal neurodevelopmental trajectory.

Journal: :Blood 2011
Jakub Tolar In-Hyun Park Lily Xia Chris J Lees Brandon Peacock Beau Webber Ron T McElmurry Cindy R Eide Paul J Orchard Michael Kyba Mark J Osborn Troy C Lund John E Wagner George Q Daley Bruce R Blazar

Mucopolysaccharidosis type I (MPS IH; Hurler syndrome) is a congenital deficiency of α-L-iduronidase, leading to lysosomal storage of glycosaminoglycans that is ultimately fatal following an insidious onset after birth. Hematopoietic cell transplantation (HCT) is a life-saving measure in MPS IH. However, because a suitable hematopoietic donor is not found for everyone, because HCT is associated...

Journal: :Acta crystallographica. Section D, Biological crystallography 2000
L Ruth D Eisenberg E F Neufeld

While seeking conditions for single crystals of human alpha-L-iduronidase, solutions were discovered (pH 3.0-8.5 containing calcium or zinc salts) that transform soluble alpha-L-iduronidase to a solid aggregate. This aggregate is a spherulite of semi-crystalline protein. The X-ray diffraction pattern and ability to bind Congo red characterize the alpha-L-iduronidase spherulite as 'amyloid-like'...

2009
Alzbeta Vazna Clare Beesley Linda Berna Larisa Stolnaja Helena Myskova Michaela Bouckova Hana Vlaskova Helena Poupetova Jiri Zeman Martin Magner Anna Hlavata Bryan Winchester Martin Hrebicek Lenka Dvorakova

Mucopolysaccharidosis type I (MPS I) is an autosomal recessive lysosomal storage disorder that is caused by a deficiency of the enzyme alpha-L-iduronidase (IDUA). Of the 21 Czech and Slovak patients who have been diagnosed with MPS I in the last 30 years, 16 have a severe clinical presentation (Hurler syndrome), 2 less severe manifestations (Scheie syndrome), and 3 an intermediate severity (Hur...

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