نتایج جستجو برای: fabry

تعداد نتایج: 6280  

2017
Michael Mauer Alexey Sokolovskiy Jay A Barth Jeffrey P Castelli Hadis N Williams Elfrida R Benjamin Behzad Najafian

OBJECTIVE Deficiency of α-galactosidase A (αGal-A) in Fabry disease leads to the accumulation mainly of globotriaosylceramide (GL3) in multiple renal cell types. Glomerular podocytes are relatively resistant to clearance of GL3 inclusions by enzyme replacement therapy (ERT). Migalastat, an orally bioavailable small molecule capable of chaperoning misfolded αGal-A to lysosomes, is approved in th...

Journal: :Human mutation 2008
Masaaki Shimotori Hiroki Maruyama Gen Nakamura Takayuki Suyama Fumiko Sakamoto Masaaki Itoh Shigeaki Miyabayashi Takahiro Ohnishi Norio Sakai Mari Wataya-Kaneda Mitsuru Kubota Toshiyuki Takahashi Tatsuhiko Mori Katsuhiko Tamura Shinji Kageyama Nobuo Shio Teruhiko Maeba Hirokazu Yahagi Motoko Tanaka Masayo Oka Hitoshi Sugiyama Toshiyuki Sugawara Noriko Mori Hiroko Tsukamoto Keiichi Tamagaki Shuuji Tanda Yuka Suzuki Chiya Shinonaga Jun-ichi Miyazaki Satoshi Ishii Fumitake Gejyo

Fabry disease is an X-linked recessive inborn metabolic disorder caused by a deficiency of the lysosomal enzyme alpha-galactosidase A (EC 3.2.1.22). The causative mutations are diverse, include both large rearrangements and single-base substitutions, and are dispersed throughout the 7 exons of the alpha-galactosidase A gene (GLA). Mutation hotspots for Fabry disease do not exist. We examined 62...

Journal: :Journal of the American Society of Nephrology : JASN 2007
Hindia Tahir Leslie L Jackson David G Warnock

This report describes an open-label, nonrandomized, prospective evaluation of the effects of angiotensin-converting enzyme inhibitor and angiotensin receptor blocker therapy on patients who have Fabry disease and also received enzyme replacement therapy with agalsidase-beta, given at 1 mg/kg body wt every 2 wk. Previous placebo-controlled phase III and phase IV trials with agalsidase-beta demon...

2015
Yeo Jin Jeon Namhee Jung Joo-Won Park Hae-Young Park Sung-Chul Jung Jung Weon Lee

Fabry disease is a lysosomal storage disorder caused by deficiency of alpha-galactosidase A (α-gal A), which results in the deposition of globotriaosylceramide (Gb3) in the vascular endothelium. Globotriaosylsphingosine (lyso-Gb3), a deacylated Gb3, is also increased in the plasma of patients with Fabry disease. Renal fibrosis is a key feature of advanced Fabry disease patients. Therefore, we e...

Journal: :Optics letters 2003
T P White L C Botten R C McPhedran C Martijn de Sterke

A novel design for an ultracompact, high-Q notch-rejection filter is presented, and an analytic expression for the transmission properties is derived. This folded directional coupler shares the properties of a Fabry-Perot resonator and a directional coupler. We compare and contrast the device to high-Q Fabry-Perot cavities in photonic crystal waveguides.

2011
U. Feldt-Rasmussen

Fabry disease, an X-linked lysosomal storage disorder, results from deficient activity of the enzyme α-galactosidase A. Affected males with the classic phoenotype have acroparaesthesias, hypohidrosis, and corneal opacities in childhood and develop renal failure, cardiac hypertrophy or strokes in the third to fifth decade of life. Some female heterozygotes are asymptomatic, some as severely affe...

2001
Pengyue Wen Michael Sanchez Matthias Gross Osman Kibar Sadik C. Esener

Two different approaches are commonly used for Fabry-Perot Semiconductor Optical Amplifiers (FP SOAs) performance analysis: the Fabry-Perot resonator approach and rate equation approach. Compared with the Fabry-Perot resonator approach, the rate equation approach is more powerful because noise and mode-related performance analysis can be included. However, it has been shown that the results bas...

2013
Langis Michaud

Purpose. To report a new clinical finding related to Fabry disease. Methods. Fabry subjects were enrolled in the study, matched for age and sex with healthy individuals as a control group. This is a prospective review of all upper lid pictures taken for every subject at their last visit. A 4-step grading scale is proposed to classify this new entity. Results. Group A (Fabry) comprised 16 males ...

2017
Sun Hee Heo Eungu Kang Yoon-Myung Kim Heounjeong Go Kyung Yong Kim Jae Yong Jung Minji Kang Gu-Hwan Kim Jae-Min Kim In-Hee Choi Jin-Ho Choi Sung-Chul Jung Robert J Desnick Han-Wook Yoo Beom Hee Lee

BACKGROUND Fabry disease is characterised by the progressive accumulation of globotriaosylceramide (Gb3) and related glycosphingolipids in vascular endothelial cells. Enzyme replacement therapy (ERT) clears this accumulation. We analysed plasma proteome profiles before and after ERT to characterise its molecular pathology. METHODS Two-dimensional electrophoresis and matrix-assisted laser deso...

Journal: :Revista portuguesa de cardiologia : orgao oficial da Sociedade Portuguesa de Cardiologia = Portuguese journal of cardiology : an official journal of the Portuguese Society of Cardiology 2014
Francisca Caetano Ana Botelho Paula Mota Joana Silva António Leitão Marques

Anderson-Fabry disease is an X-linked lysosomal storage disorder caused by abnormalities of the GLA gene, which encodes the enzyme α-galactosidase A. A deficiency of this enzyme leads to the lysosomal accumulation of glycosphingolipids, which may cause left ventricular hypertrophy that is typically concentric and symmetric. We present the case of a 60-year-old woman with symptoms of dyspnea, at...

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