Literature DB >> 293739

Analysis of genetic complementation by whole-cell microtechniques in fibroblast heterokaryons.

R A Gravel, A Leung, M Saunders, P Hösli.   

Abstract

A whole-cell microtechnique for the determination of complementation of human metabolic disorders is presented. This procedure permits the isolation of individual multinucleate cells produced by cell fusion for the quantitative evaluation of complementation. Mutant fibroblasts with a deficiency of propionyl-CoA carboxylase activity (EC 6.4.1.3) that had been mapped to complementation groups pcc and bio were used to evaluate the microtechnique. Complementation was monitored by the determination of [14C]propionate incorporation into cellular macromolecules. Single cells or a small number of cells were isolated from plastic film dishes after radioactive incubation by cutting out the portion of the plastic film holding the desired cells. Isotope incorporation was linear in 10-50 unfused cells and in 10-50 fused normal cells containing five or more nuclei. There was also a direct correlation between the nuclear content of cells and the amount of isotope incorporated. Three pcc and two bio mutants were fused in pairwise combinations by means of polyethylene glycol and complementation was determined by isotope incorporation in sets of 50 multinucleate cells, each cell isolated individually. The results agreed with autoradiographic data for both complementing and noncomplementing strains. The method is quantitative and gives severalfold higher sensitivity than current procedures. The method can be applied to the complementation analysis of a wide variety of inherited disorders of intermediary metabolism.

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Year:  1979        PMID: 293739      PMCID: PMC411897          DOI: 10.1073/pnas.76.12.6520

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  21 in total

1.  Genetic complementation of propionyl-CoA carboxylase deficiency in cultured human fibroblasts.

Authors:  R A Gravel; K F Lam; K J Scully; Y Hsia
Journal:  Am J Hum Genet       Date:  1977-07       Impact factor: 11.025

2.  Quantitative assays of enzyme activity in single cells: early prenatal diagnosis of genetic disorders.

Authors:  P Hösli
Journal:  Clin Chem       Date:  1977-08       Impact factor: 8.327

3.  Genetic complementation among inherited deficiencies of methylmalonyl-CoA mutase activity: evidence for a new class of human cobalamin mutant.

Authors:  H F Willard; I S Mellman; L E Rosenberg
Journal:  Am J Hum Genet       Date:  1978-01       Impact factor: 11.025

4.  Biochemical differences between mutant propionyl-CoA carboxylases from two complementation groups.

Authors:  B Wolf; Y E Hsia; L E Rosenberg
Journal:  Am J Hum Genet       Date:  1978-09       Impact factor: 11.025

5.  Rapid prenatal and postnatal detection of inborn errors of propionate, methylmalonate, and cobalamin metabolism: a sensitive assay using cultured cells.

Authors:  H F Willard; L M Ambani; A C Hart; M J Mahoney; L E Rosenberg
Journal:  Hum Genet       Date:  1976-12-15       Impact factor: 4.132

6.  Biotin-response organicaciduria. Multiple carboxylase defects and complementation studies with propionicacidemia in cultured fibroblasts.

Authors:  M Saunders; L Sweetman; B Robinson; K Roth; R Cohn; R A Gravel
Journal:  J Clin Invest       Date:  1979-12       Impact factor: 14.808

7.  Infantile sialidosis: a phenocopy of type 1 GM1 gangliosidosis distinguished by genetic complementation and urinary oligosaccharides.

Authors:  R A Gravel; J A Lowden; J W Callahan; L S Wolfe; N M Ng Yin Kin
Journal:  Am J Hum Genet       Date:  1979-11       Impact factor: 11.025

8.  Polyethylene glycol-induced mammalian cell hybridization: effect of polyethylene glycol molecular weight and concentration.

Authors:  R L Davidson; K A O'Malley; T B Wheeler
Journal:  Somatic Cell Genet       Date:  1976-05

9.  Juvenile Sandhoff Disease: complementation tests with Sandhoff and Tay-Sachs disease using polyethylene glycol-induced cell fusion.

Authors:  S Wood
Journal:  Hum Genet       Date:  1978-04-24       Impact factor: 4.132

10.  Plaque formation and isolation of pure lines with poliomyelitis viruses.

Authors:  R DULBECCO; M VOGT
Journal:  J Exp Med       Date:  1954-02       Impact factor: 14.307

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  4 in total

1.  Complementation analysis in Gaucher disease using single cell microassay techniques. Evidence for a single "Gaucher gene".

Authors:  R A Gravel; A Leung
Journal:  Hum Genet       Date:  1983       Impact factor: 4.132

2.  Ultramicro assays of enzymes for rapid and inexpensive diagnosis of inherited disorders of metabolism.

Authors:  L Sweetman
Journal:  Indian J Pediatr       Date:  1981 Mar-Apr       Impact factor: 1.967

3.  Hunter disease (mucopolysaccharidosis type II) associated with unbalanced inactivation of the X chromosomes in a karyotypically normal girl.

Authors:  J T Clarke; W L Greer; P M Strasberg; R D Pearce; M A Skomorowski; P N Ray
Journal:  Am J Hum Genet       Date:  1991-08       Impact factor: 11.025

4.  Studies on complementation in argininosuccinate synthetase and argininosuccinate lyase deficiencies in human fibroblasts.

Authors:  L Cathelineau; D Pham Dinh; P Briand; P Kamoun
Journal:  Hum Genet       Date:  1981       Impact factor: 4.132

  4 in total

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