نتایج جستجو برای: atp7b

تعداد نتایج: 482  

Journal: :The Journal of biological chemistry 2007
Dominik Huster Tina D Purnat Jason L Burkhead Martina Ralle Oliver Fiehn Franziska Stuckert N Erik Olson Daniel Teupser Svetlana Lutsenko

Copper is essential for human physiology, but in excess it causes the severe metabolic disorder Wilson disease. Elevated copper is thought to induce pathological changes in tissues by stimulating the production of reactive oxygen species that damage multiple cell targets. To better understand the molecular basis of this disease, we performed genome-wide mRNA profiling as well as protein and met...

2016
Malgorzata Lenartowicz Torben Moos Mateusz Ogórek Thomas G. Jensen Lisbeth B. Møller

Deficiency of one of the copper transporters ATP7A and ATP7B leads to the rare X-linked disorder Menkes Disease (MD) or the rare autosomal disorder Wilson disease (WD), respectively. In order to investigate whether the ATP7A and the ATP7B genes may be transcriptionally regulated, we measured the expression level of the two genes at various concentrations of iron, copper, and insulin. Treating f...

Journal: :Journal of clinical and diagnostic research : JCDR 2013
Mani Kant Kumar Vijay Kumar Praphul Kumar Singh

Wilson's Disease (WD) is a rare, autosomal, recessive, inborn error of the copper metabolism, which is caused by a mutation in the copper-transporting gene, ATP7B. The presentation is usually neurologic or hepatic, which is seen in 40% of the patients. The diagnosis depends primarily on the clinical features, the biochemical parameters and the presence of the Kayser - Fleischer ring. Here, we a...

2017
Giusy Ranucci Roman Polishchuck Raffaele Iorio

Wilson's disease (WD) is an autosomal recessive disorder of copper metabolism, caused by mutations in the ATP7B gene. A clear demand for novel WD treatment strategies has emerged. Although therapies using zinc salts and copper chelators can effectively cure WD, these drugs exhibit limitations in a substantial pool of WD patients who develop intolerance and/or severe side effects. Several lines ...

Journal: :American journal of physiology. Regulatory, integrative and comparative physiology 2006
Shannon L Kelleher Bo Lönnerdal

Milk copper (Cu) concentration declines and directly reflects the stage of lactation. Three Cu-specific transporters (Ctr1, Atp7A, Atp7B) have been identified in the mammary gland; however, the integrated role they play in milk Cu secretion is not understood. Whereas the regulation of milk composition by the lactogenic hormone prolactin (PRL) has been documented, the specific contribution of PR...

Journal: :Journal of Gastroenterology and Hepatology 2004

Journal: :The Journal of clinical investigation 2016
Josef Lichtmannegger Christin Leitzinger Ralf Wimmer Sabine Schmitt Sabine Schulz Yaschar Kabiri Carola Eberhagen Tamara Rieder Dirk Janik Frauke Neff Beate K Straub Peter Schirmacher Alan A DiSpirito Nathan Bandow Bipin S Baral Andrew Flatley Elisabeth Kremmer Gerald Denk Florian P Reiter Simon Hohenester Friedericke Eckardt-Schupp Norbert A Dencher Jerzy Adamski Vanessa Sauer Christoph Niemietz Hartmut H J Schmidt Uta Merle Daniel Nils Gotthardt Guido Kroemer Karl Heinz Weiss Hans Zischka

In Wilson disease (WD), functional loss of ATPase copper-transporting β (ATP7B) impairs biliary copper excretion, leading to excessive copper accumulation in the liver and fulminant hepatitis. Current US Food and Drug Administration- and European Medicines Agency-approved pharmacological treatments usually fail to restore copper homeostasis in patients with WD who have progressed to acute liver...

2016
Dániel Németh Kristóf Árvai Péter Horváth János Pál Kósa Bálint Tobiás Bernadett Balla Anikó Folhoffer Anna Krolopp Péter András Lakatos Ferenc Szalay

Objective. Wilson's disease is a disorder of copper metabolism which is fatal without treatment. The great number of disease-causing ATP7B gene mutations and the variable clinical presentation of WD may cause a real diagnostic challenge. The emergence of next-generation sequencing provides a time-saving, cost-effective method for full sequencing of the whole ATP7B gene compared to the tradition...

Journal: :The Journal of clinical investigation 2015
Clavia Ruth Wooton-Kee Ajay K Jain Martin Wagner Michael A Grusak Milton J Finegold Svetlana Lutsenko David D Moore

Wilson's disease (WD) is an autosomal recessive disorder that results in accumulation of copper in the liver as a consequence of mutations in the gene encoding the copper-transporting P-type ATPase (ATP7B). WD is a chronic liver disorder, and individuals with the disease present with a variety of complications, including steatosis, cholestasis, cirrhosis, and liver failure. Similar to patients ...

Journal: :Hepatology 2005
Aftab Ala Jimo Borjigin Arnold Rochwarger Michael Schilsky

Wilson Disease (WD) usually presents in the first decades of life, although rare patients have a later presentation. We report the clinical features, diagnostic evaluation, and outcome with treatment of two septuagenarian siblings evaluated as part of a research trial for treatment of neurological WD. The index case was a 72-year-old woman who suffered progressive neurological disability, then ...

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