Literature DB >> 6766899

Hybridization studies of fibroblasts from Hurler, Scheie, and Hurler/Scheie compound patients: support for the hypothesis of allelic mutants.

J J Fortuin, W J Kleijer.   

Abstract

Heterokaryons were made by fusion of alpha-L-iduronidase-deficient fibroblasts from patients with the Hurler, Scheie, or Hurler/Scheie compound syndrome. The fused cell populations remained alpha-L-iduronidase deficient and accumulated 35S-labeled glycosaminoglycans (35S-GAG) to the same extent as the parental cells strains. Also, when 35S-GAG accumulation was studied by autoradiography at the level of single bi- and multinuclear hybrid cells, no evidence was found for genetic complementation. The results support the hypothesis that the mutations in the Hurler and Scheie syndromes are allelic, and they are compatible with the view that patients with intermediate phenotypes represent genetic compounds.

Entities:  

Mesh:

Substances:

Year:  1980        PMID: 6766899     DOI: 10.1007/bf00273487

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


  18 in total

1.  Genetic complementation after fusion of Tay-Sachs and Sandhoff cells.

Authors:  G H Thomas; H A Taylor; C S Miller; J Axelman; B R Migeon
Journal:  Nature       Date:  1974-08-16       Impact factor: 49.962

2.  Tay-Sachs and Sandhoff's disease: intergenic complementation after somatic cell hybridization.

Authors:  H Galjaard; A Hoogeveen; H A de Wit-Verbeek; A J Reuser; W Keijzer; A Westerveld; D Bootsma
Journal:  Exp Cell Res       Date:  1974-08       Impact factor: 3.905

3.  Allelism, non-allelism, and genetic compounds among the mucopolysaccharidoses.

Authors:  V A McKusick; R R Howell; I E Hussels; E F Neufeld; R E Stevenson
Journal:  Lancet       Date:  1972-05-06       Impact factor: 79.321

4.  The Hurler corrective factor. Purification and some properties.

Authors:  R W Barton; E F Neufeld
Journal:  J Biol Chem       Date:  1971-12-25       Impact factor: 5.157

5.  Scheie and Hurler syndromes: apparent identity of the biochemical defect.

Authors:  U Wiesmann; E F Neufeld
Journal:  Science       Date:  1970-07-03       Impact factor: 47.728

6.  Rapid prenatal diagnosis of GM1-gangliosidosis using microchemical methods.

Authors:  W J Kleijer; E Van der Veer; M F Niermeijer
Journal:  Hum Genet       Date:  1976-08-30       Impact factor: 4.132

7.  Alpha-L-iduronidase activity in cultured skin fibroblasts and amniotic fluid cells.

Authors:  C W Hall; E F Neufeld
Journal:  Arch Biochem Biophys       Date:  1973-10       Impact factor: 4.013

8.  A method for alpha-L-iduronidase assay.

Authors:  J Singh; P Niebes; N Di Ferrante
Journal:  FEBS Lett       Date:  1974-09-01       Impact factor: 4.124

9.  The iduronidase-deficient mucopolysaccharidoses: clinical and roentgenorgraphic features.

Authors:  R E Stevenson; R R Howell; V A McKusick; R Suskind; J W Hanson; D E Elliott; E F Neufeld
Journal:  Pediatrics       Date:  1976-01       Impact factor: 7.124

10.  The defect in the Hurler and Scheie syndromes: deficiency of -L-iduronidase.

Authors:  G Bach; R Friedman; B Weissmann; E F Neufeld
Journal:  Proc Natl Acad Sci U S A       Date:  1972-08       Impact factor: 11.205

View more
  5 in total

1.  Lack of complementation in somatic cell hybrids between fibroblasts from patients with different forms of cystinosis.

Authors:  O L Pellett; M L Smith; A A Greene; J A Schneider
Journal:  Proc Natl Acad Sci U S A       Date:  1988-05       Impact factor: 11.205

2.  Genetic complementation analysis in somatic cell hybrids of alpha-L-iduronidase deficient cells.

Authors:  M Wehnert; G Machill; L Petruschka
Journal:  Hum Genet       Date:  1985       Impact factor: 4.132

3.  Computed tomography studies on patients with mucopolysaccharidoses.

Authors:  R W Watts; E Spellacy; B E Kendall; G du Boulay; D A Gibbs
Journal:  Neuroradiology       Date:  1981-02       Impact factor: 2.804

4.  Properties of alpha-L-iduronidase in cultured skin fibroblasts from alpha-L-iduronidase-deficient patients.

Authors:  S Fujibayashi; R Minami; Y Ishikawa; K Wagatsuma; T Nakao; S Tsugawa
Journal:  Hum Genet       Date:  1984       Impact factor: 4.132

5.  Genetic heterogeneity of Fanconi's anemia demonstrated by somatic cell hybrids.

Authors:  S Zakrzewski; K Sperling
Journal:  Hum Genet       Date:  1980       Impact factor: 4.132

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.