Abstract
The primary structure of polycystin predicts a large integral membrane protein with multiple cell recognition motifs, but its function remains unknown. Insight into polycystin's normal function and its role in the development of autosomal dominant polycystic kidney disease (PKD1) requires the assembly of an extensive collection of molecular reagents to examine its expression and create model systems for functional studies. Development of these crucial reagents has been complicated due to the presence of transcriptionally active homologous loci. We have assembled the authentic full-length PKD1 cDNA and demonstrated expression of polycystin in vitro. Polyclonal antibodies directed against distinct extra- and intracellular domains specifically immunoprecipitated in vitro translated polycystin. The panel of antibodies was used to determine localization of polycystin in renal epithelial and endothelial cell lines and tissues of fetal, adult, and cystic origins. In normal adult kidney and maturing fetal nephrons, polycystin expression was confined to epithelial cells of the distal nephron and vascular endothelial cells. Expression in the proximal nephron was only observed after injury-induced cell proliferation. Polycystin expression was confined to ductal epithelium in liver, pancreas, and breast, and restricted to astrocytes in normal brain. We report clear evidence for the membrane localization of polycystin by both tissue sections and by confocal microscopy in cultured renal and endothelial cells. Interestingly, when cultured cells made cell-cell contact, polycystin was localized to the lateral membranes of cells in contact. These data suggest that polycystin is likely to have a widespread role in epithelial cell differentiation and maturation and in cell-cell interactions.
MeSH Terms
Adult
Brain/embryology,metabolism
Cell Line
Cells, Cultured
DNA, Complementary
Endothelium, Vascular/metabolism
Epithelium/metabolism
Fetus
Gene Library
Humans
Kidney/metabolism
Nephrons/embryology,metabolism
Organ Specificity
Polycystic Kidney, Autosomal Dominant
Polymerase Chain Reaction
Protein Biosynthesis
Proteins/chemistry
Recombinant Proteins/biosynthesis,chemistry
Subcellular Fractions/metabolism
TRPP Cation Channels
Chemicals
DNA, Complementary
Proteins
Recombinant Proteins
TRPP Cation Channels
polycystic kidney disease 1 protein
Authors & Affiliations
19 authors, click to expand affiliations / ORCID
Ibraghimov-Beskrovnaya O
Genzyme Genetics, P.O. Box 9322, Framingham, MA 01701-9322, USA.
Dackowski W R
Foggensteiner L
Coleman N
Thiru S
Petry L R
Burn T C
Connors T D
Van Raay T
Bradley J
Qian F
Onuchic L F
Watnick T J
Piontek K
Hakim R M
Landes G M
Germino G G
Sandford R
Klinger K W
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