The DOI matches its Crossref record; 2 of the 2 references it cites were found in a registry or an index.
Biochemical Characterization of the Wilson Disease Protein and Functional Expression in the Yeast Saccharomyces cerevisiae
Open accessAbstract
Wilson disease is a disorder of copper metabolism characterized by hepatic cirrhosis and neuronal degeneration due to inherited mutations in a gene encoding a putative copper-transporting P-type ATPase. Polyclonal antisera generated against the amino terminus of the Wilson protein detected a specific 165-kDa protein in HepG2 and CaCo cell lysates. Further analysis revealed that this protein is synthesized as a single-chain polypeptide and localized to the trans-Golgi network under steady state conditions. An increase in the copper concentration resulted in the rapid movement of this protein to a cytoplasmic vesicular compartment. This copper-specific cellular redistribution of the Wilson protein is a reversible process that occurs independent of a new protein synthesis. Expression of the wild-type but not mutant Wilson protein in the ccc2Delta strain of Saccharomyces cerevisiae restored copper incorporation into the multicopper oxidase Fet3p, providing direct evidence of copper transport by the Wilson protein. Taken together these data reveal a remarkable evolutionary conservation in the cellular mechanisms of copper metabolism and provide a unique model for the regulation of copper transport into the secretory pathway of eucaryotic cells.
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Cites
Molecular mechanisms of CFTR chloride channel dysfunction in cystic fibrosis
Welsh, M. J., & Smith, A. E. (1993) · Cell · doi:10.1016/0092-8674(93)90353-r · Cited by 1,485
The FET3 gene of S. cerevisiae encodes a multicopper oxidase required for ferrous iron uptake.
Askwith, C., Eide, D., Van Ho, A., Bernard, P. S., Li, L., Davis-Kaplan, S., Sipe, D. M., & Kaplan, J. (1994) · Cell · doi:10.1016/0092-8674(94)90346-8 · Cited by 670
Cited by
Function and Regulation of Human Copper-Transporting ATPases
Lutsenko, S., Barnes, N. L., Bartee, M. Y., & Dmitriev, O. Y. (2007) · Physiological Reviews · doi:10.1152/physrev.00004.2006 · Cited by 864
Liver cell death and anemia in Wilson disease involve acid sphingomyelinase and ceramide
Lang, P. A., Schenck, M., Nicolay, J. P., Becker, J. U., Kempe, D. S., Lupescu, A., Koka, S., Eisele, K., Klarl, B. A., Rübben, H., et al. (2007) · Nature Medicine · doi:10.1038/nm1539 · Cited by 438
Stern, B. R., Solioz, M., Krewski, D., Aggett, P., Aw, T.-C., Baker, S., Crump, K., Dourson, M., Haber, L., Hertzberg, R., et al. (2007) · Journal of Toxicology and Environmental Health, Part B · doi:10.1080/10937400600755911 · Cited by 373
Essential role for Atox1 in the copper-mediated intracellular trafficking of the Menkes ATPase
Hamza, I., Prohaska, J., & Gitlin, J. D. (2003) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0336230100 · Cited by 203
Self-citationCu(I) binding and transfer by the N terminus of the Wilson disease protein.
Yatsunyk, L. A., & Rosenzweig, A. C. (2007) · Journal of Biological Chemistry · doi:10.1074/jbc.m609533200 · Cited by 130
Metadata
- Authors
- Hung, I. H., Suzuki, M., Yamaguchi, Y., Yuan, D. S., Klausner, R. D., & Gitlin, J. D.
- Venue
- Journal of Biological Chemistry
- Publisher
- Elsevier BV
- Published
- 1997-08
- Volume
- 272
- Issue
- 34
- Pages
- 21461-21466
- ISSN
- 0021-9258 (ISSN-L)
- OpenAlex
- W2047390883
- Cited by
- 324
- References
- 2
- FWCI
- 25.47
- Type
- journal article
- Language
- en
- Open access
- hybrid
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- Hung, I. H., Suzuki, M., Yamaguchi, Y., Yuan, D. S., Klausner, R. D., & Gitlin, J. D. (1997). Biochemical Characterization of the Wilson Disease Protein and Functional Expression in the Yeast Saccharomyces cerevisiae. Journal of Biological Chemistry. https://doi.org/10.1074/jbc.272.34.21461
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