نتایج جستجو برای: i mucopolysaccharidosis i

تعداد نتایج: 1040570  

Journal: :Acta Scientiae Veterinariae 2021

2017
Rossella Parini Federica Deodato Maja Di Rocco Edoardo Lanino Franco Locatelli Chiara Messina Attilio Rovelli Maurizio Scarpa

Mucopolysaccharidosis I-Hurler (MPS I-H) is the most severe form of a metabolic genetic disease caused by mutations of IDUA gene encoding the lysosomal α-L-iduronidase enzyme. MPS I-H is a rare, life-threatening disease, evolving in multisystem morbidity including progressive neurological disease, upper airway obstruction, skeletal deformity and cardiomyopathy. Allogeneic hematopoietic stem cel...

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: :PloS one 2016
Omar Khalid Moin U Vera Philip L Gordts N Matthew Ellinwood Philip H Schwartz Patricia I Dickson Jeffrey D Esko Raymond Y Wang

BACKGROUND Cardiovascular disease, a progressive manifestation of α-L-iduronidase deficiency or mucopolysaccharidosis type I, continues in patients both untreated and treated with hematopoietic stem cell transplantation or intravenous enzyme replacement. Few studies have examined the effects of α-L-iduronidase deficiency and subsequent glycosaminoglycan storage upon arterial gene expression to ...

Journal: :Genetics and molecular research : GMR 2007
R P El Dib G M Pastores

Mucopolysaccharidoses are a group of inherited metabolic diseases caused by the absence or deficiency of the lysosomal enzymes that are needed for breaking down glycosaminoglycans (GAGs). Over time, GAGs collect in cells, blood and connective tissues, and increased amounts are excreted in the urine. The result is permanent and includes progressive cell damage that affects the individual's appea...

2016
Meena Bolourchi Pierangelo Renella Raymond Y. Wang

The prevalence of aortic root dilatation (ARD) in mucopolysaccharidosis (MPS) is not well documented. We investigated aortic root measurements in 34 MPS patients at the Children's Hospital of Orange County (CHOC). The diagnosis, treatment status, age, gender, height, weight and aortic root parameters (aortic valve annulus (AVA), sinuses of Valsalva (SoV), and sinotubular junction (STJ)) were ex...

Journal: :The American journal of pathology 1983
M E Haskins G D Aguirre P F Jezyk R J Desnick D F Patterson

Five cats with feline alpha-L-iduronidase-deficient mucopolysaccharidosis were studied. Membrane-bound cytoplasmic inclusions were present in central nervous system neurons, hepatocytes, chondrocytes, vascular and splenic smooth muscle cells, bone marrow leukocytes, and fibroblasts of the skin, eye, and cardiac valves. The lesions in these cats closely resemble those described in human patients...

Journal: :The Journal of Bone and Joint Surgery. British volume 1996

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