نتایج جستجو برای: mucopolysaccharidosis 1

تعداد نتایج: 2754438  

Journal: :Human mutation 2011
Francesca Bertola Mirella Filocamo Giorgio Casati Matthew Mort Camillo Rosano Anna Tylki-Szymanska Beyhan Tüysüz Orazio Gabrielli Serena Grossi Maurizio Scarpa Giancarlo Parenti Daniela Antuzzi Jaime Dalmau Maja Di Rocco Carlo Dionisi Vici Ilyas Okur Jordi Rosell Attilio Rovelli Francesca Furlan Miriam Rigoldi Andrea Biondi David N Cooper Rossella Parini

Mutational analysis of the IDUA gene was performed in a cohort of 102 European patients with mucopolysaccharidosis type I. A total of 54 distinct mutant IDUA alleles were identified, 34 of which were novel including 12 missense mutations, 2 nonsense mutations, 12 splicing mutations, 5 micro-deletions, 1 micro-duplication 1 translational initiation site mutation, and 1 'no-stop' change (p.X654Re...

Journal: :Journal of Inborn Errors of Metabolism and Screening 2015

Journal: :PathoGenetics 2009
Alessandra Tessitore Marinella Pirozzi Alberto Auricchio

BACKGROUND Lysosomal storage diseases are characterized by intracellular accumulation of metabolites within lysosomes. Recent evidence suggests that lysosomal storage impairs autophagy resulting in accumulation of polyubiquitinated proteins and dysfunctional mitochondria, ultimately leading to apoptosis. We studied the relationship between lysosome storage and impairment of different intracellu...

Journal: :The Journal of the Association of Physicians of India 2013
Rajesh Patil Nilesh Wasekar S G Jadhav Ravindra Zore Parin Sangoi Deepti Vishwanathan

+Pg Resident, *Head of unit, **assistant Professor, Dept. of Medicine, grant Medical College and sir J.J. group of Hospitals, Mumbai. Received: 01.10.2011; Revised: 16.07.2012; Re-revised: 03.12.2012; accepted: 11.01.2013 Abstract Introduction : We present a very rare case of mucopolysaccharidosis type II (Hunter syndrome). which presented as short stature, coarse facies, mild mental retardatio...

ژورنال: افق دانش 2020

Aims Morquio syndrome is a mucopolysaccharidosis (type 4) that has autosomal recessive inheritance. Moreover, it is caused by defects in the two genes; GALNS (Murcio A) and GLB1 (Murcio B). The prevalence rate of this condition is estimated to be about 1 per 200000 live births globally. Besides, Middle Eastern cases shape the greatest ratio, due to higher rates of consanguineous marriages. The ...

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...

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: :Archives of ophthalmology 2007
Susanne Pitz Olufunmilola Ogun Manal Bajbouj Laila Arash Gudrun Schulze-Frenking Michael Beck

OBJECTIVE To describe the progression of ocular changes in patients with mucopolysaccharidosis I receiving enzyme replacement therapy. METHODS Three male and five female patients with mucopolysaccharidosis I were followed up for 4 years while undergoing enzyme replacement therapy with alpha-l-iduronidase (Aldurazyme). Visual acuity, corneal clouding, intraocular pressure, ophthalmoscopy, and ...

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 ...

نمودار تعداد نتایج جستجو در هر سال

با کلیک روی نمودار نتایج را به سال انتشار فیلتر کنید