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PMID: 7507932 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

Stoichiometry of recombinant cystic fibrosis transmembrane conductance regulator in epithelial cells and its functional reconstitution into cells in vitro.

The Journal of biological chemistry ·Vol. 269 ·No. 4 ·1994-01-28 ·Pages 2987-95

Marshall J, Fang S, Ostedgaard LS, O'Riordan CR, Ferrara D, Amara JF, Hoppe H, Scheule RK, Welsh MJ, Smith AE

Abstract

We have generated several clones of Chinese hamster ovary, mouse epitheloid C127, and pig kidney epithelial LLCPK1 cells producing high levels of functional recombinant human cystic fibrosis transmembrane conductance regulator (CFTR). Processing of CFTR to the mature and fully glycosylated form in these cells is inefficient with only approximately 40% of all newly synthesized CFTR being converted to the mature form. Furthermore, expression of the most frequent mutant allele of the cystic fibrosis (CF) gene, the delta F508 mutant in these epithelial cells, indicated that it is biosynthetically arrested at the endoplasmic reticulum and fails to traffic to the plasma membrane. Using a combination of CFTR mutants and monoclonal antibodies, all the detectable recombinant CFTR in these cells was determined at least under the conditions used, to be present as a monomer. To demonstrate the feasibility of protein replacement therapy, we were able to effect the physical transfer of functional recombinant CFTR produced in Chinese hamster ovary cells to the plasma membranes of Ha3b fibroblasts, a cell line devoid of cAMP-stimulated chloride channels. Transfer of CFTR was mediated by the hemagglutinin viral fusion protein of influenza virus present on the Ha3b cells. Efficiency of transfer was up to 25% of the target cells, and CFTR chloride channel activity was detectable for up to 12 h post-fusion. Therefore, with the development of an appropriate formulation of fusogenic proteoliposome or virosome containing reconstituted purified CFTR, it should be feasible to introduce functional CFTR into CF-affected cells.

MeSH Terms
Adenosine Triphosphate/metabolism Animals CHO Cells Cell Line Cricetinae Cyclic AMP-Dependent Protein Kinases/metabolism Cystic Fibrosis/metabolism Cystic Fibrosis Transmembrane Conductance Regulator Electrophoresis, Polyacrylamide Gel Epithelium/metabolism Genetic Vectors Humans Kidney Kinetics Membrane Fusion Membrane Proteins/analysis,biosynthesis,metabolism Methionine/metabolism Mice Mutagenesis, Site-Directed Phosphorylation Rats Recombinant Proteins/analysis,biosynthesis,metabolism Restriction Mapping Sulfur Radioisotopes Swine Thyroid Gland/metabolism Time Factors Transfection
Chemicals
CFTR protein, human Membrane Proteins Recombinant Proteins Sulfur Radioisotopes Cystic Fibrosis Transmembrane Conductance Regulator Adenosine Triphosphate Methionine Cyclic AMP-Dependent Protein Kinases
Authors & Affiliations
10 authors, click to expand affiliations / ORCID
Marshall J
Genzyme Corporation, Framingham, Massachusetts 01701.
Fang S
Ostedgaard L S
O'Riordan C R
Ferrara D
Amara J F
Hoppe H
Scheule R K
Welsh M J
Smith A E
Article Info
Journal
The Journal of biological chemistry
Abbr.
J Biol Chem
ISSN
0021-9258
Published
1994-01-28
Pages
2987-95
Language
English
Region
United States
NLM ID
2985121R
Subset
IM
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