Literature DB >> 11854914

Copper transportion of WD protein in hepatocytes from Wilson disease patients in vitro.

G Q Hou1, X L Liang, R Chen, L W Tang, Y Wang, P Y Xu, Y R Zhang, C H Ou.   

Abstract

AIM: To study the effect of copper transporting P-type ATPase in copper metabolism of hepatocyte and pathogenesis of Wilson disease (WD).
METHODS: WD copper transporting properties in some organelles of the cultured hepatocytes were studied from WD patients and normal controls.These cultured hepatocytes were incubated in the media of copper 15 mg x L(-1) only, copper 15 mg x L(-1) with vincristine (agonist of P-type ATPase) 0.5mg x L(-1), or copper 15 mg x L(-1) with vanadate (antagonist of P-type ATPase) 18.39 mg x L(-1) separately. Microsome (endoplasmic reticulum and Golgi apparatus), lysosome, mitochondria, and cytosol were isolated by differential centrifugation. Copper contents in these organelles were measured with atomic absorption spectrophotometer, and the influence in copper transportion of these organelles by vanadate and vincristine were comparatively analyzed between WD patients and controls. WD copper transporting P-type ATPase was detected by SDS-PAGE in conjunction with Western blot in liver samples of WD patients and controls.
RESULTS: The specific WD proteins (M(r)155,000 lanes) were expressed in human hepatocytes, including the control and WD patients. After incubation with medium containing copper for 2 h or 24 h, the microsome copper concentration in WD patients was obviously lower than that of controls, and the addition of vanadate or vincristine would change the copper transporting of microsomes obviously. When incubated with vincristine, levels of copper in microsome were significantly increased, while incubated with vanadate, the copper concentrations in microsome were obviously decreased. The results indicated that there were WD proteins, the copper transportion P-type ATPase in the microsome of hepatocytes. WD patients possessed abnormal copper transporting function of WD protein in the microsome, and the agonist might correct the defect of copper transportion by promoting the activity of copper transportion P-type ATPase.
CONCLUSION: Copper transportion P-type ATPase plays an important role in hepatocytic copper metabolism. Dysfunction of hepatocytic WD protein copper transportion might be one of the most important factors for WD.

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Year:  2001        PMID: 11854914      PMCID: PMC4695607          DOI: 10.3748/wjg.v7.i6.846

Source DB:  PubMed          Journal:  World J Gastroenterol        ISSN: 1007-9327            Impact factor:   5.742


  42 in total

1.  Molecular analysis and diagnosis in Japanese patients with Wilson's disease.

Authors:  N Shimizu; H Nakazono; Y Takeshita; C Ikeda; H Fujii; A Watanabe; Y Yamaguchi; H Hemmi; H Shimatake; T Aoki
Journal:  Pediatr Int       Date:  1999-08       Impact factor: 1.524

2.  [Study on mutation of exon 8 of Wilson's disease gene].

Authors:  P Xu; X Liang; S Ma
Journal:  Zhonghua Yi Xue Yi Chuan Xue Za Zhi       Date:  1999-04

3.  Identification and analysis of mutations in the Wilson disease gene (ATP7B): population frequencies, genotype-phenotype correlation, and functional analyses.

Authors:  A B Shah; I Chernov; H T Zhang; B M Ross; K Das; S Lutsenko; E Parano; L Pavone; O Evgrafov; I A Ivanova-Smolenskaya; G Annerén; K Westermark; F H Urrutia; G K Penchaszadeh; I Sternlieb; I H Scheinberg; T C Gilliam; K Petrukhin
Journal:  Am J Hum Genet       Date:  1997-08       Impact factor: 11.025

4.  Comparison of long lasting therapeutic effects between succimer and penicillamine on hepatolenticular degeneration.

Authors:  Ming-Shan Ren; Zhi Zhang; Jun-Xia Wu; Fei Li; Ben-Chun Xue; Ren-Min Yang
Journal:  World J Gastroenterol       Date:  1998-12       Impact factor: 5.742

5.  Bile acid formation in primary human hepatocytes.

Authors:  Curt Einarsson; Ewa Ellis; Anna Abrahamsson; Bo-Goran Ericzon; Ingermar Bjorkhem; Magnus Axelson
Journal:  World J Gastroenterol       Date:  2000-08       Impact factor: 5.742

Review 6.  ATP7B (WND) protein.

Authors:  K Terada; M L Schilsky; N Miura; T Sugiyama
Journal:  Int J Biochem Cell Biol       Date:  1998-10       Impact factor: 5.085

Review 7.  Physiologic function of the Wilson disease gene product, ATP7B.

Authors:  M J Bingham; T J Ong; K H Summer; R B Middleton; H J McArdle
Journal:  Am J Clin Nutr       Date:  1998-05       Impact factor: 7.045

8.  Genetic expression of Wilson's disease in cell culture: a diagnostic marker.

Authors:  W Y Chan; W Cushing; M A Coffman; O M Rennert
Journal:  Science       Date:  1980-04-18       Impact factor: 47.728

9.  Restoration of holoceruloplasmin synthesis in LEC rat after infusion of recombinant adenovirus bearing WND cDNA.

Authors:  K Terada; T Nakako; X L Yang; M Iida; N Aiba; Y Minamiya; M Nakai; T Sakaki; N Miura; T Sugiyama
Journal:  J Biol Chem       Date:  1998-01-16       Impact factor: 5.157

10.  Functional characterization of missense mutations in ATP7B: Wilson disease mutation or normal variant?

Authors:  J R Forbes; D W Cox
Journal:  Am J Hum Genet       Date:  1998-12       Impact factor: 11.025

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