Literature DB >> 6885075

Detection of the carrier state for an X-linked disorder, the Lesch-Nyhan syndrome, by the use of lymphocyte cloning.

J L Dempsey, A A Morley, R S Seshadri, B T Emmerson, R Gordon, C I Bhagat.   

Abstract

Using a limiting dilution technique, we found that the frequency of thioguanine resistant (TGR) lymphocyte clones was less than 5.0 X 10(-5) in 14 normal individuals, between 9.0 X 10(-3) and 8.9 X 10(-2) in seven heterozygotes for Lesch-Nyhan syndrome, and 0.88 and 0.87 in two hemizygotes. TGR clones from heterozygotes were expanded and had the hemizygote phenotype as evidenced by low hypoxanthine incorporation and severely deficient hypoxanthine-guanine-phosphoribosyl-transferase activity. Enumeration of TGR lymphocyte clones provides a simple technique for detection of heterozygosity for Lesch-Nyhan syndrome. A similar approach using lymphocyte cloning may be suitable for detection of the carrier state for other X-linked disorders.

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Year:  1983        PMID: 6885075     DOI: 10.1007/bf00279414

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  10 in total

1.  Selective in vitro growth of T lymphocytes from normal human bone marrows.

Authors:  D A Morgan; F W Ruscetti; R Gallo
Journal:  Science       Date:  1976-09-10       Impact factor: 47.728

2.  Cloning of human lymphocytes using limiting dilution.

Authors:  A A Morley; K J Trainor; R S Seshadri
Journal:  Exp Hematol       Date:  1983-05       Impact factor: 3.084

3.  Evidence that thioguanine-resistant lymphocytes detected by autoradiography are mutant cells.

Authors:  J L Dempsey; A A Morley
Journal:  Mutat Res       Date:  1983-02       Impact factor: 2.433

4.  Measurement of in vivo mutations in human lymphocytes.

Authors:  A A Morley; K J Trainor; R Seshadri; R G Ryall
Journal:  Nature       Date:  1983-03-10       Impact factor: 49.962

5.  Evidence for the derivation of individual hair roots from three progenitor cells.

Authors:  J Dancis; D N Silvers; M E Balis; R P Cox; M S Schwartz
Journal:  Hum Genet       Date:  1981       Impact factor: 4.132

6.  Lesch-Nyhan syndrome: rapid detection of heterozygotes by use of hair follicles.

Authors:  S M Gartler; R C Scott; J L Goldstein; B Campbell
Journal:  Science       Date:  1971-05-07       Impact factor: 47.728

7.  Studies on the coordinate activity and liability of orotidylate phosphoribosyltransferase and decarboxylase in human erythrocytes, and the effects of allopurinol administration.

Authors:  R M Fox; M H Wood; W J O'Sullivan
Journal:  J Clin Invest       Date:  1971-05       Impact factor: 14.808

8.  An enumerative assay of purine analogue resistant lymphocytes in women heterozygous for the Lesch-Nyhan Mutation.

Authors:  G H Strauss; E F Allen; R J Albertini
Journal:  Biochem Genet       Date:  1980-06       Impact factor: 1.890

9.  Detection of Lesch-Nyhan syndrome carriers: analysis of hair roots for HPRT by agarose gel electrophoresis and autoradiography.

Authors:  B Bakay; C Tucker-Pian; J E Seegmiller
Journal:  Clin Genet       Date:  1980-06       Impact factor: 4.438

10.  Enumeration of thioguanine-resistant lymphocytes using autoradiography.

Authors:  A A Morley; S Cox; D Wigmore; R Seshadri; J L Dempsey
Journal:  Mutat Res       Date:  1982-08       Impact factor: 2.433

  10 in total
  7 in total

1.  Gender-specific frequency of background somatic mutations at the hypoxanthine phosphoribosyltransferase locus in cord blood T lymphocytes from preterm newborns.

Authors:  M Yoshioka; P M Vacek; T Poseno; R Silver; B A Finette
Journal:  Proc Natl Acad Sci U S A       Date:  1999-01-19       Impact factor: 11.205

2.  Mutation rates at the glycophorin A and HPRT loci in uranium miners exposed to radon progeny.

Authors:  E M Shanahan; D Peterson; D Roxby; J Quintana; A A Morely; A Woodward
Journal:  Occup Environ Med       Date:  1996-07       Impact factor: 4.402

3.  Spontaneous 6-thioguanine-resistant lymphocytes in Fanconi anemia patients and their heterozygous parents.

Authors:  E Wunder; T M Schroeder
Journal:  Hum Genet       Date:  1985       Impact factor: 4.132

4.  Molecular basis of hypoxanthine-guanine phosphoribosyltransferase deficiency in Italian Lesch-Nyhan patients: identification of nine novel mutations.

Authors:  M Bertelli; D Randi; V Micheli; S Gallo; G Andrighetto; P Parmigiani; G Jacomelli; M Carella; C Lievore; M Pandolfo
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

5.  Selection against blood cells deficient in hypoxanthine phosphoribosyltransferase (HPRT) in Lesch-Nyhan heterozygotes occurs at the level of multipotent stem cells.

Authors:  M Hakoda; Y Hirai; M Akiyama; H Yamanaka; C Terai; N Kamatani; S Kashiwazaki
Journal:  Hum Genet       Date:  1995-12       Impact factor: 4.132

6.  Increased frequency of 6-thioguanine-resistant peripheral blood lymphocytes in Werner syndrome patients.

Authors:  K Fukuchi; K Tanaka; Y Kumahara; K Marumo; M B Pride; G M Martin; R J Monnat
Journal:  Hum Genet       Date:  1990-02       Impact factor: 4.132

7.  Tankyrase inhibitor XAV-939 enhances osteoblastogenesis and mineralization of human skeletal (mesenchymal) stem cells.

Authors:  Nuha Almasoud; Nihal AlMuraikhi; Sarah Binhamdan; Ghaydaa Younis; Hanouf Alaskar; Amal Alotaibi; Muthurangan Manikandan; Musaad Alfayez; Moustapha Kassem
Journal:  Sci Rep       Date:  2020-10-07       Impact factor: 4.379

  7 in total

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