Literature DB >> 809585

Variability of acid hydrolase activities in cultured skin fibroblasts and amniotic fluid cells.

E Young, P Willcox, A E Whitfield, A D Patrick.   

Abstract

The specific activities of lysosomal hydrolases in cultured skin fibroblasts and amniotic fluid cells showed wide and unpredictable variations between cultures, which may lead to difficulty in differentiating normal, heterozygous, and homozygous cells. However, the variability for a given culture was similar for all enzymes assayed, so that a clearer differentiation of a relative deficiency of a given enzyme could be obtained by expressing its activity in ratio to that of another enzyme. Activity ratios were particularly useful in the evaluation of enzyme levels in cultured amniotic fluid cells. Results of their application to tests of pregnancies at risk for metachromatic leucodystrophy, Krabbe's leucodystrophy, GM1-gangliosidosis, and GM2-gangliosidosis (Sandhoff variant) are presented.

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Year:  1975        PMID: 809585      PMCID: PMC1013282          DOI: 10.1136/jmg.12.3.224

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  21 in total

1.  Protein measurement with the Folin phenol reagent.

Authors:  O H LOWRY; N J ROSEBROUGH; A L FARR; R J RANDALL
Journal:  J Biol Chem       Date:  1951-11       Impact factor: 5.157

2.  Letter: Prenatal diagnosis of Tay-Sachs disease.

Authors:  R B Ellis; J U Ikonne; A D Patrick; R Stephens; P Willcox
Journal:  Lancet       Date:  1973-11-17       Impact factor: 79.321

3.  Late onset GM2-gangliosidosis. Clinical, pathological, and biochemical studies on 8 patients.

Authors:  E M Brett; R B Ellis; L Haas; J U Ikonne; B D Lake; A D Patrick; R Stephens
Journal:  Arch Dis Child       Date:  1973-10       Impact factor: 3.791

4.  [Effect of different sera on mucopolysaccharide synthesis in fibroblast cultures (author's transl)].

Authors:  P D Kittlick; G Neupert; U Lümkemann
Journal:  Exp Pathol (Jena)       Date:  1973

5.  Infantile metachromatic leukodystrophy.

Authors:  M M Kaback; R R Howell
Journal:  N Engl J Med       Date:  1970-06-11       Impact factor: 91.245

6.  Effect of culture conditions on enzyme activities in cultivated human fibroblasts.

Authors:  C A Ryan; S Y Lee; H L Nadler
Journal:  Exp Cell Res       Date:  1972       Impact factor: 3.905

7.  Deficiency of arylsulphatase A in leucocytes and skin fibroblasts in juvenile machromatic leucodystrophy.

Authors:  J G Leroy; J Dumon; J Radermecker
Journal:  Nature       Date:  1970-05-09       Impact factor: 49.962

8.  Beta-glucosidase activity in fibroblasts from homozygotes and heterozygotes for Gaucher's disease.

Authors:  E Beutler; W Kuhl; F Trinidad; R Teplitz; H Nadler
Journal:  Am J Hum Genet       Date:  1971-01       Impact factor: 11.025

9.  Ganglioside GM2 storage diseases: hexosaminidase deficiencies in cultured fibroblasts.

Authors:  S Okada; M L Veath; J Leroy; J S O'Brien
Journal:  Am J Hum Genet       Date:  1971-01       Impact factor: 11.025

10.  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

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  7 in total

1.  Prenatal diagnosis of Hurler disease by analysis of alpha-iduronidase in chorionic villi.

Authors:  E P Young
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

2.  Culture conditions found to minimize false positive diagnosis of lysosomal storage disorders.

Authors:  J Arnon; A Ornoy; G Bach
Journal:  In Vitro Cell Dev Biol       Date:  1988-12

3.  The effects of sucrose loading on lysosomal hydrolases.

Authors:  T Kato; S Okada; T Yutaka; H Yabuuchi
Journal:  Mol Cell Biochem       Date:  1984       Impact factor: 3.396

4.  Juvenile Gaucher's disease with horizontal gaze palsy in three siblings.

Authors:  J H Tripp; B D Lake; E Young; J Ngu; E M Brett
Journal:  J Neurol Neurosurg Psychiatry       Date:  1977-05       Impact factor: 10.154

5.  The use of natural and artifical substrates in the prenatal diagnosis of Krabbe's disease.

Authors:  G T Besley
Journal:  J Inherit Metab Dis       Date:  1978       Impact factor: 4.982

6.  Treatment of HL-60 cells with dimethyl sulphoxide inhibits the formation of beta-N-acetylhexosaminidase S.

Authors:  C Emiliani; F Falzetti; A Orlacchio; J L Stirling
Journal:  Biochem J       Date:  1990-11-15       Impact factor: 3.857

7.  Neuraminidase deficiency: case report and review of the phenotype.

Authors:  I D Young; E P Young; J Mossman; A R Fielder; J R Moore
Journal:  J Med Genet       Date:  1987-05       Impact factor: 6.318

  7 in total

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