نتایج جستجو برای: mucopolysaccharidosis type i

تعداد نتایج: 2218703  

Journal: :Neurology 2016
Wladimir Bocca Vieira de Rezende Pinto Paulo Victor Sgobbi de Souza Gabriel Novaes de Rezende Batistella Acary Souza Bulle Oliveira

A 2-year-old boy presented with delayed motor skills and language since birth. Family history disclosed consanguineous parents. Examination showed global muscular hypotonia, optic atrophy, oculomotor apraxia, and normal head circumference. Brain MRI showed optic atrophy and macrocerebellum (figure). Laboratory tests revealed deficient activity of a-L-iduronidase in peripheral blood leukocytes a...

Journal: :Molecular genetics and metabolism 2007
N Matthew Ellinwood Marie-Anne Colle Margaret A Weil Margret L Casal Charles H Vite Staci Wiemelt Christopher W Hasson Thomas M O'Malley Xingxuan He Ulana Prociuk Lucie Verot John R Melniczek Anne Lannon Gustavo D Aguirre Van W Knox Sydney M Evans Marie T Vanier Edward H Schuchman Steven U Walkley Mark E Haskins

Severe mucopolysaccharidosis type I (MPS I) is a fatal neuropathic lysosomal storage disorder with significant skeletal involvement. Treatment involves bone marrow transplantation (BMT), and although effective, is suboptimal, due to treatment sequelae and residual disease. Improved approaches will need to be tested in animal models and compared to BMT. Herein we report on bone marrow transplant...

Journal: :مجله دانشگاه علوم پزشکی شهید صدوقی یزد 0
منصور صالحی m salehi . [email protected] رسول صالحی r salehi بهرام نصر اصفهانی b nasr- esfahani

introduction: mucopolysaccharidosis i (mps-i) is an autosomal recessive lysosomal storage diseases, caused by α-l-iduronidase (idua) enzyme deficiency. the clinical manifestations of mps-i patients are variable ranging from severe to mild, and therefore prediction of disease severity is difficult. from when idua gene has been cloned more than 109 distinct mutations have been identified in it an...

2013
Elena L. Aronovich Bryan C. Hall Jason B. Bell R. Scott McIvor Perry B. Hackett

The Sleeping Beauty transposon system, a non-viral, integrating vector that can deliver the alpha-L-iduronidase-encoding gene, is efficient in correcting mucopolysaccharidosis type I in NOD/SCID mice. However, in previous studies we failed to attain reliable long-term alpha-L-iduronidase expression in immunocompetent mice. Here, we focused on achieving sustained high-level expression in immunoc...

2014
Violetta Opoka-Winiarska

Introduction One of the most important manifestations of mucopolysaccharidosis (MPS) type I, II and VI is a progressive disease of the osteoarticular system. The evaluation of the disease advancement is difficult due to the complexity of symptoms. The characteristic features are progressive limitation of joint mobility and joint pain. These symptoms affect the quality of patient life. A uniform...

2010
Tsing-San Hsu

Correspondence should be addressed to Tsing-San Hsu, [email protected] Received 8 September 2010; Accepted 18 October 2010 Academic Editor: Julio Rossi Copyright q 2010 Tsing-San Hsu. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly c...

Journal: :Heart 1999
T A Fischer H A Lehr U Nixdorff J Meyer

The genetic mucopolysaccharidosis syndromes (MPS) are autosomal recessive inborn errors of metabolism. Heart valve involvement in MPS is not uncommon but only a few case reports of successful cardiac surgery are available. In particular, reports of combined aortic and mitral stenosis associated with MPS type I-S are very rare. Both type I and type VI MPS are associated with significant left sid...

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