The DOI matches its Crossref record; 21 of the 21 references it cites were found in a registry or an index.
Polycystin-2, the protein mutated in autosomal dominant polycystic kidney disease (ADPKD), is a Ca 2+ -permeable nonselective cation channel
Open accessAbstract
Defects in polycystin-2, a ubiquitous transmembrane glycoprotein of unknown function, is a major cause of autosomal dominant polycystic kidney disease (ADPKD), whose manifestation entails the development of fluid-filled cysts in target organs. Here, we demonstrate that polycystin-2 is present in term human syncytiotrophoblast, where it behaves as a nonselective cation channel. Lipid bilayer reconstitution of polycystin-2-positive human syncytiotrophoblast apical membranes displayed a nonselective cation channel with multiple subconductance states, and a high perm-selectivity to Ca 2+ . This channel was inhibited by anti-polycystin-2 antibody, Ca 2+ , La 3+ , Gd 3+ , and the diuretic amiloride. Channel function by polycystin-2 was confirmed by patch-clamping experiments of polycystin-2 heterologously infected Sf9 insect cells. Further, purified insect cell-derived recombinant polycystin-2 and in vitro translated human polycystin-2 had similar ion channel activity. The polycystin-2 channel may be associated with fluid accumulation and/or ion transport regulation in target epithelia, including placenta. Dysregulation of this channel provides a mechanism for the onset and progression of ADPKD.
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Cites
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Cited by
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Autosomal dominant polycystic kidney disease
Torres, V. E., Harris, P. C., & Pirson, Y. (2007) · The Lancet · doi:10.1016/s0140-6736(07)60601-1 · Cited by 1,550
Self-citationThe Ciliopathies: An Emerging Class of Human Genetic Disorders
Badano, J. L., Mitsuma, N., Beales, P. L., & Katsanis, N. (2006) · Annual Review of Genomics and Human Genetics · doi:10.1146/annurev.genom.7.080505.115610 · Cited by 1,144
The TRP Superfamily of Cation Channels
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Metadata
- Authors
- González-Perrett, S., Kim, K., Ibarra, C., Damiano, A. E., Zotta, E., Batelli, M., Harris, P. C., Reisin, I. L., Arnaout, M. A., & Cantiello, H. F.
- Venue
- Proceedings of the National Academy of Sciences
- Published
- 2000-12-26
- Volume
- 98
- Issue
- 3
- Pages
- 1182-1187
- ISSN
- 0027-8424 (ISSN-L)
- 1091-6490
- OpenAlex
- W2014264816
- Cited by
- 447
- References
- 21
- FWCI
- 13.73
- Type
- journal article
- Language
- en
- Open access
- bronze
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- González-Perrett, S., Kim, K., Ibarra, C., Damiano, A. E., Zotta, E., Batelli, M., Harris, P. C., Reisin, I. L., Arnaout, M. A., & Cantiello, H. F. (2001). Polycystin-2, the protein mutated in autosomal dominant polycystic kidney disease (ADPKD), is a Ca 2+ -permeable nonselective cation channel. Proceedings of the National Academy of Sciences. https://doi.org/10.1073/pnas.98.3.1182
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