Literature DB >> 9950803

Genetic disorders of membrane transport. IV. Wilson's disease and Menkes disease.

M Schaefer1, J D Gitlin.   

Abstract

Copper is an essential transition metal that permits the facile transfer of electrons in a series of critical biochemical pathways. Menkes disease and Wilson's disease are inherited disorders of copper metabolism resulting from the absence or dysfunction of homologous copper-transporting ATPases that reside in the trans-Golgi network of all cells. Despite striking differences in the clinical presentation of these two diseases, the respective ATPases function in precisely the same manner within the cell and the unique clinical features of each disease are entirely the result of the tissue-specific expression of each protein. Elucidation of the basic defect in these rare genetic disorders has provided a valuable heuristic paradigm for understanding the mechanisms of cellular copper homeostasis.

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Year:  1999        PMID: 9950803     DOI: 10.1152/ajpgi.1999.276.2.G311

Source DB:  PubMed          Journal:  Am J Physiol        ISSN: 0002-9513


  21 in total

1.  The molecular and genetic base of congenital transport defects.

Authors:  J F Desjeux
Journal:  Gut       Date:  2000-05       Impact factor: 23.059

2.  Systems biology approach in Chlamydomonas reveals connections between copper nutrition and multiple metabolic steps.

Authors:  Madeli Castruita; David Casero; Steven J Karpowicz; Janette Kropat; Astrid Vieler; Scott I Hsieh; Weihong Yan; Shawn Cokus; Joseph A Loo; Christoph Benning; Matteo Pellegrini; Sabeeha S Merchant
Journal:  Plant Cell       Date:  2011-04-15       Impact factor: 11.277

Review 3.  Cellular multitasking: the dual role of human Cu-ATPases in cofactor delivery and intracellular copper balance.

Authors:  Svetlana Lutsenko; Arnab Gupta; Jason L Burkhead; Vesna Zuzel
Journal:  Arch Biochem Biophys       Date:  2008-05-21       Impact factor: 4.013

4.  Structure-function analysis of purified Enterococcus hirae CopB copper ATPase: effect of Menkes/Wilson disease mutation homologues.

Authors:  K D Bissig; H Wunderli-Ye; P W Duda; M Solioz
Journal:  Biochem J       Date:  2001-07-01       Impact factor: 3.857

5.  Physical characterization of high-affinity gastrointestinal Cu transport in vitro in freshwater rainbow trout Oncorhynchus mykiss.

Authors:  Sunita R Nadella; Martin Grosell; Chris M Wood
Journal:  J Comp Physiol B       Date:  2006-07-12       Impact factor: 2.200

6.  Role of the vagus in the reduced pancreatic exocrine function in copper-deficient rats.

Authors:  Tanja Babic; Ruchi Bhagat; Shuxia Wan; Kirsteen N Browning; Michael Snyder; Samuel R Fortna; R Alberto Travagli
Journal:  Am J Physiol Gastrointest Liver Physiol       Date:  2012-12-28       Impact factor: 4.052

Review 7.  Copper chaperones for cytochrome c oxidase and human disease.

Authors:  Iqbal Hamza; Jonathan D Gitlin
Journal:  J Bioenerg Biomembr       Date:  2002-10       Impact factor: 2.945

8.  Eukaryotic CTR copper uptake transporters require two faces of the third transmembrane domain for helix packing, oligomerization, and function.

Authors:  Stephen G Aller; Edward T Eng; Christopher J De Feo; Vinzenz M Unger
Journal:  J Biol Chem       Date:  2004-09-22       Impact factor: 5.157

9.  Participation of ATP7A in macrophage mediated oxidation of LDL.

Authors:  Zhenyu Qin; Eddy S Konaniah; Bonnie Neltner; Raphael A Nemenoff; David Y Hui; Neal L Weintraub
Journal:  J Lipid Res       Date:  2009-11-23       Impact factor: 5.922

10.  Analysis of the human homologue of the canine copper toxicosis gene MURR1 in Wilson disease patients.

Authors:  Bettina Stuehler; Juergen Reichert; Wolfgang Stremmel; Mark Schaefer
Journal:  J Mol Med (Berl)       Date:  2004-06-17       Impact factor: 4.599

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