Literature DB >> 9053545

Immunoblot analysis of peroxisomal proteins in liver and fibroblasts from patients.

R J Wanders1, C Dekker, R Ofman, R B Schutgens, P Mooijer.   

Abstract

Identification of a patient as suffering from a peroxisomal disorder usually starts by the finding of elevated very long-chain fatty acids in plasma and/or serum. This is followed by more detailed studies in blood, fibroblasts and tissues, including immunoblot analysis. Indeed, immunoblot analysis has become a valuable tool in the correct diagnosis and assignment of individual patients, except for X-linked adrenoleukodystrophy (X-ALD). We describe a simple immunoblotting procedure applicable to liver and fibroblast homo-genates using antibodies raised against catalase and the three beta-oxidation enzyme proteins acyl-CoA oxidase I, bifunctional protein and peroxisomal thiolase. The same procedure can also be used for chorionic villus biopsy specimens and has now become the method of choice for the prenatal diagnosis of Zellweger syndrome (and other disorders of peroxisome biogenesis) and rhizomelic chondrodysplasia punctata.

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Year:  1995        PMID: 9053545     DOI: 10.1007/bf00711433

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  42 in total

1.  Identification of pristanoyl-CoA oxidase as a distinct, clofibrate non-inducible enzyme in rat liver peroxisomes.

Authors:  R J Wanders; S Denis; C Jakobs; H J ten Brink
Journal:  Biochim Biophys Acta       Date:  1992-03-04

2.  First prenatal diagnosis of acyl-CoA oxidase deficiency.

Authors:  R J Wanders; A Schelen; N Feller; R B Schutgens; F Stellaard; C Jakobs; B Mitulla; G Seidlitz
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

3.  Heterogeneity of catalase staining in human hepatocellular peroxisomes.

Authors:  F Roels; A Cornelis
Journal:  J Histochem Cytochem       Date:  1989-03       Impact factor: 2.479

4.  Pathology of hepatic peroxisomes and mitochondria in patients with peroxisomal disorders.

Authors:  J L Hughes; A Poulos; E Robertson; C W Chow; L J Sheffield; J Christodoulou; R F Carter
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1990

5.  Measurement of peroxisomal fatty acid beta-oxidation in cultured human skin fibroblasts.

Authors:  R J Wanders; S Denis; J P Ruiter; R B Schutgens; C W van Roermund; B S Jacobs
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

Review 6.  Human liver pathology in peroxisomal diseases: a review including novel data.

Authors:  F Roels; M Espeel; F Poggi; H Mandel; L van Maldergem; J M Saudubray
Journal:  Biochimie       Date:  1993       Impact factor: 4.079

7.  Purification and properties of acyl-CoA oxidase from rat liver.

Authors:  T Osumi; T Hashimoto; N Ui
Journal:  J Biochem       Date:  1980-06       Impact factor: 3.387

8.  Zellweger-like syndrome with detectable hepatic peroxisomes: a variant form of peroxisomal disorder.

Authors:  Y Suzuki; N Shimozawa; T Orii; N Igarashi; N Kono; A Matsui; Y Inoue; S Yokota; T Hashimoto
Journal:  J Pediatr       Date:  1988-11       Impact factor: 4.406

9.  Peroxisomal bifunctional enzyme deficiency.

Authors:  P A Watkins; W W Chen; C J Harris; G Hoefler; S Hoefler; D C Blake; A Balfe; R I Kelley; A B Moser; M E Beard
Journal:  J Clin Invest       Date:  1989-03       Impact factor: 14.808

10.  Cytoplasmic catalase and ghostlike peroxisomes in the liver from a child with atypical chondrodysplasia punctata.

Authors:  M Espeel; J C Heikoop; J A Smeitink; F A Beemer; D De Craemer; M Van den Berg; T Hashimoto; R J Wanders; R B Schutgens; B T Poll-The
Journal:  Ultrastruct Pathol       Date:  1993 Nov-Dec       Impact factor: 1.094

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  9 in total

1.  A new peroxisomal beta-oxidation disorder in twin neonates: defective oxidation of both cerotic and pristanic acids.

Authors:  E Christensen; S A Pedersen; H Leth; C Jakobs; R B Schutgens; R J Wanders
Journal:  J Inherit Metab Dis       Date:  1997-09       Impact factor: 4.982

2.  A novel PEX12 mutation identified as the cause of a peroxisomal biogenesis disorder with mild clinical phenotype, mild biochemical abnormalities in fibroblasts and a mosaic catalase immunofluorescence pattern, even at 40 degrees C.

Authors:  Avraham Zeharia; Merel S Ebberink; Ronald J A Wanders; Hans R Waterham; Alisa Gutman; Andreea Nissenkorn; Stanley H Korman
Journal:  J Hum Genet       Date:  2007-05-30       Impact factor: 3.172

3.  Immunocytochemical localization of peroxisomal proteins in human liver and kidney.

Authors:  M Espeel; G Van Limbergen
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

4.  Allelic Expression Imbalance Promoting a Mutant PEX6 Allele Causes Zellweger Spectrum Disorder.

Authors:  Kim D Falkenberg; Nancy E Braverman; Ann B Moser; Steven J Steinberg; Femke C C Klouwer; Agatha Schlüter; Montserrat Ruiz; Aurora Pujol; Martin Engvall; Karin Naess; FrancJan van Spronsen; Irene Körver-Keularts; M Estela Rubio-Gozalbo; Sacha Ferdinandusse; Ronald J A Wanders; Hans R Waterham
Journal:  Am J Hum Genet       Date:  2017-12-07       Impact factor: 11.025

5.  A PEX6-defective peroxisomal biogenesis disorder with severe phenotype in an infant, versus mild phenotype resembling Usher syndrome in the affected parents.

Authors:  Annick Raas-Rothschild; Ronald J A Wanders; Petra A W Mooijer; Jeannette Gootjes; Hans R Waterham; Alisa Gutman; Yasuyuki Suzuki; Nobuyuki Shimozawa; Naomi Kondo; Gideon Eshel; Marc Espeel; Frank Roels; Stanley H Korman
Journal:  Am J Hum Genet       Date:  2002-02-28       Impact factor: 11.025

6.  Isolated dihydroxyacetonephosphate-acyl-transferase deficiency in rhizomelic chondrodysplasia punctata: clinical presentation, metabolic and histological findings.

Authors:  H Hebestreit; R J Wanders; R B Schutgens; M Espeel; I Kerckaert; F Roels; B Schmausser; L Schrod; A Marx
Journal:  Eur J Pediatr       Date:  1996-12       Impact factor: 3.183

7.  2-Methyl-3-hydroxybutyryl-CoA dehydrogenase deficiency is caused by mutations in the HADH2 gene.

Authors:  Rob Ofman; Jos P N Ruiter; Marike Feenstra; Marinus Duran; Bwee Tien Poll-The; Johannes Zschocke; Regina Ensenauer; Willy Lehnert; Jörn Oliver Sass; Wolfgang Sperl; Ronald J A Wanders
Journal:  Am J Hum Genet       Date:  2003-04-14       Impact factor: 11.025

8.  Arginine improves peroxisome functioning in cells from patients with a mild peroxisome biogenesis disorder.

Authors:  Kevin Berendse; Merel S Ebberink; Lodewijk Ijlst; Bwee Tien Poll-The; Ronald J A Wanders; Hans R Waterham
Journal:  Orphanet J Rare Dis       Date:  2013-09-09       Impact factor: 4.123

Review 9.  The important role of biochemical and functional studies in the diagnostics of peroxisomal disorders.

Authors:  Sacha Ferdinandusse; Merel S Ebberink; Frédéric M Vaz; Hans R Waterham; Ronald J A Wanders
Journal:  J Inherit Metab Dis       Date:  2016-03-04       Impact factor: 4.982

  9 in total

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