Literature DB >> 3732317

A prenatal test for the cerebro-hepato-renal (Zellweger) syndrome by demonstration of the absence of catalase-containing particles (peroxisomes) in cultured amniotic fluid cells.

R J Wanders, G Schrakamp, H van den Bosch, J M Tager, R B Schutgens.   

Abstract

In this paper we show that whereas acyl-CoA: dihydroxyacetone phosphate acyltransferase, a membrane-bound peroxisomal enzyme, is deficient in homogenates of cultured amniotic fluid cells of fetuses with Zellweger syndrome, catalase a soluble peroxisomal matrix enzyme is present in normal amounts. Digitonin titration experiments revealed a striking difference in the percentage of particle-bound catalase in control and Zellweger amniocytes: in Zellweger amniocytes all catalase activity was found to be present in the soluble cytoplasm, (less than 5% particle-bound), whereas in control amniocytes catalase was found to be predominantly particle-bound (62% +/- 8%, n = 5). Measurement of the percentage of particle-bound catalase by means of digitonin titrations thus provides a simple prenatal test for Zellweger syndrome via the direct demonstration of the presence or absence of catalase-containing particles (peroxisomes).

Entities:  

Mesh:

Substances:

Year:  1986        PMID: 3732317     DOI: 10.1007/bf00441876

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  13 in total

1.  Cerebro-hepato-renal syndrome of Zellweger. A report of eight cases with comments upon the incidence, the liver lesion, and a fault in pipecolic acid metabolism.

Authors:  D M Danks; P Tippett; C Adams; P Campbell
Journal:  J Pediatr       Date:  1975-03       Impact factor: 4.406

2.  Prenatal detection of Zellweger syndrome.

Authors:  R B Schutgens; H S Heymans; R J Wanders; H vd Bosch; G Schrakamp
Journal:  Lancet       Date:  1984-12-08       Impact factor: 79.321

3.  Properties of guinea pig liver peroxisomal dihydroxyacetone phosphate acyltransferase.

Authors:  C L Jones; A K Hajra
Journal:  J Biol Chem       Date:  1980-09-10       Impact factor: 5.157

4.  Cerebro-hepato-renal (Zellweger) syndrome and neonatal adrenoleukodystrophy: similarities in phenotype and accumulation of very long chain fatty acids.

Authors:  F R Brown; A J McAdams; J W Cummins; R Konkol; I Singh; A B Moser; H W Moser
Journal:  Johns Hopkins Med J       Date:  1982-12

5.  Deficiency of acyl-CoA: dihydroxyacetone phosphate acyltransferase in patients with Zellweger (cerebro-hepato-renal) syndrome.

Authors:  R B Schutgens; G J Romeyn; R J Wanders; H van den Bosch; G Schrakamp; H S Heymans
Journal:  Biochem Biophys Res Commun       Date:  1984-04-16       Impact factor: 3.575

6.  Activity of peroxisomal enzymes and intracellular distribution of catalase in Zellweger syndrome.

Authors:  R J Wanders; M Kos; B Roest; A J Meijer; G Schrakamp; H S Heymans; W H Tegelaers; H van den Bosch; R B Schutgens; J M Tager
Journal:  Biochem Biophys Res Commun       Date:  1984-09-28       Impact factor: 3.575

7.  Cyanide-insensitive and clofibrate enhanced beta-oxidation of dodecanedioic acid in rat liver. An indication of peroxisomal beta-oxidation of N-dicarboxylic acids.

Authors:  P B Mortensen; S Kølvraa; N Gregersen; K Rasmussen
Journal:  Biochim Biophys Acta       Date:  1982-11-12

8.  Formation of cholic acid from 3 alpha, 7 alpha, 12 alpha-trihydroxy-5 beta-cholestanoic acid by rat liver peroxisomes.

Authors:  F Kase; I Björkhem; J I Pedersen
Journal:  J Lipid Res       Date:  1983-12       Impact factor: 5.922

9.  Lignoceric acid is oxidized in the peroxisome: implications for the Zellweger cerebro-hepato-renal syndrome and adrenoleukodystrophy.

Authors:  I Singh; A E Moser; S Goldfischer; H W Moser
Journal:  Proc Natl Acad Sci U S A       Date:  1984-07       Impact factor: 11.205

10.  Subcellular localization of acyl coenzyme A: dihydroxyacetone phosphate acyltransferase in rat liver peroxisomes (microbodies).

Authors:  A K Hajra; C L Burke; C L Jones
Journal:  J Biol Chem       Date:  1979-11-10       Impact factor: 5.157

View more
  7 in total

1.  Complementation study of peroxisome-deficient disorders by immunofluorescence staining and characterization of fused cells.

Authors:  S Yajima; Y Suzuki; N Shimozawa; S Yamaguchi; T Orii; Y Fujiki; T Osumi; T Hashimoto; H W Moser
Journal:  Hum Genet       Date:  1992-03       Impact factor: 4.132

Review 2.  Prenatal and perinatal diagnosis of peroxisomal disorders.

Authors:  R B Schutgens; G Schrakamp; R J Wanders; H S Heymans; J M Tager; H van den Bosch
Journal:  J Inherit Metab Dis       Date:  1989       Impact factor: 4.982

3.  Infantile Refsum disease: an inherited peroxisomal disorder. Comparison with Zellweger syndrome and neonatal adrenoleukodystrophy.

Authors:  B T Poll-The; J M Saudubray; H A Ogier; M Odièvre; J M Scotto; L Monnens; L C Govaerts; F Roels; A Cornelis; R B Schutgens
Journal:  Eur J Pediatr       Date:  1987-09       Impact factor: 3.183

Review 4.  Peroxisomal disorders: a newly recognised group of genetic diseases.

Authors:  R B Schutgens; H S Heymans; R J Wanders; H van den Bosch; J M Tager
Journal:  Eur J Pediatr       Date:  1986-02       Impact factor: 3.183

Review 5.  Zellweger syndrome: biochemical procedures in diagnosis, prevention and treatment.

Authors:  R B Schutgens; R J Wanders; H S Heymans; A W Schram; J M Tager; G Schrakamp; H van den Bosch
Journal:  J Inherit Metab Dis       Date:  1987       Impact factor: 4.982

Review 6.  Prenatal diagnosis of inborn errors of metabolism with renal manifestations.

Authors:  E Harms
Journal:  Pediatr Nephrol       Date:  1987-07       Impact factor: 3.714

7.  Chondrodysplasia punctata with a mild clinical course.

Authors:  J M Nuoffer; J P Pfammatter; A Spahr; H Toplak; R J Wanders; R B Schutgens; U N Wiesmann
Journal:  J Inherit Metab Dis       Date:  1994       Impact factor: 4.982

  7 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.