نتایج جستجو برای: mucopolysaccharidosis

تعداد نتایج: 2370  

2018
Gé-Ann Kuiper Olga L. M. Meijer Eveline J. Langereis Frits A. Wijburg

BACKGROUND Rare diseases are often un- or misdiagnosed for extended periods, resulting in a long diagnostic delay that may significantly add to the burden of the disease. An early diagnosis is particularly essential if a disease-modifying treatment is available. The purpose of this study was to assess the extent of the diagnostic delay in the two ultra-rare diseases, i.e., mucopolysaccharidosis...

Journal: :The Biochemical journal 1985
J J Hopwood H Elliott

Sulphated N-acetylhexosamines have been isolated from human urine and tentatively identified as N-acetylglucosamine 6-sulphate (GlcNAc6S), N-acetylgalactosamine 6-sulphate (GalNAc6S), N-acetylgalactosamine 4-sulphate (GalNAc4S) and N-acetylgalactosamine 4,6-disulphate (GalNAc4,6diS). Urine from mucopolysaccharidosis-Type-IIID, -IVA and -VI patients compared with that from normal individuals con...

Journal: :Journal of Biological Chemistry 1996

Journal: :International Bulletin of Otorhinolaryngology 2019

Journal: :International Archives of Otorhinolaryngology 2020

Journal: :World journal of pediatrics : WJP 2015
Agnieszka Różdżyńska-Świątkowska Agnieszka Jurecka Joachim Cieślik Anna Tylki-Szymańska

BACKGROUND Mucopolysaccharidosis (MPS) diseases lead to a profound disruption in normal mechanisms of growth and development. This study was undertaken to determine the general growth of children with MPS I and II. METHODS The anthropometric data of patients with MPS I and II (n=76) were retrospectively analyzed. The growth patterns of these patients were analyzed and then plotted onto Polish...

Journal: :Clinical science 1979
J J Hopwood V Muller A C Pollard

1. a-L-Iduronidase activity was assayed by incubation of iduronosyl anhydro[ l-3H]mannitol 6-sulphate with homogenates of cultured skin fibroblasts, amniotic cells and leucocytes derived from normal individuals, patients affected with GLiduronidase deficiency disorder (mucopolysaccharidosis type I: Hurler, Scheie and Hurler-Scheie compound) and parents of such patients. 2. The assay for cc-L-id...

2010
Lucio Nitsch Francesca Cecere Paola Di Natale

Pag. 1 Background 2 1. Lysosomal storage disease 2 Pathogenetic events in lysosomal storage diseases 2 New therapeutic options for lysosomal storage diseases 5 Gene therapy by retroviral vectors 10 2. Mucopolysaccharidosis IIIB 14 Aims of the PhD thesis 16 Matherials and Methods 17 Chemicals 17 Animals 17 Western Blotting 17 Primary cortical neuronal cultures 18 Confocal immunofluorescence anal...

2017
Arunabha Ghosh Elsa Shapiro Stewart Rust Kathleen Delaney Samantha Parker Adam J Shaywitz Adelaida Morte Gillian Bubb Maureen Cleary Tien Bo Christine Lavery Brian W Bigger Simon A Jones

BACKGROUND Mucopolysaccharidosis type III is a progressive, neurodegenerative lysosomal storage disorder for which there is currently no effective therapy. Though numerous potential therapies are in development, there are several challenges to conducting clinical research in this area. We seek to make recommendations on the approach to clinical research in MPS III, including the selection of ou...

Journal: :Brain : a journal of neurology 2015
Carla Martins Helena Hůlková Larbi Dridi Virginie Dormoy-Raclet Lubov Grigoryeva Yoo Choi Alexander Langford-Smith Fiona L Wilkinson Kazuhiro Ohmi Graziella DiCristo Edith Hamel Jerôme Ausseil David Cheillan Alain Moreau Eva Svobodová Zuzana Hájková Markéta Tesařová Hana Hansíková Brian W Bigger Martin Hrebícek Alexey V Pshezhetsky

Severe progressive neurological paediatric disease mucopolysaccharidosis III type C is caused by mutations in the HGSNAT gene leading to deficiency of acetyl-CoA: α-glucosaminide N-acetyltransferase involved in the lysosomal catabolism of heparan sulphate. To understand the pathophysiology of the disease we generated a mouse model of mucopolysaccharidosis III type C by germline inactivation of ...

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