Literature DB >> 2430454

Hepatic peroxisomes are deficient in infantile refsum disease: a cytochemical study of 4 cases.

F Roels, A Cornelis, B T Poll-The, P Aubourg, H Ogier, J Scotto, J M Saudubray.   

Abstract

We examined liver biopsies from 4 patients with the infantile form of Refsum disease. No peroxisomes were visualized by light microscopy after cytochemical staining for catalase, a marker enzyme for this organelle. Absence of peroxisomes was confirmed by electron microscopy in 3 patients; in the 4th patient we observed organelles of peculiar size and structure and with minimal catalase activity. Light microscopy also showed birefringent macrophages containing P.A.S.-positive material; they were abundant in the 3 older children, and rare in the youngest (8 months). Peroxisomes and birefringent macrophages were absent in 2 patients with the cerebrohepatorenal syndrome of Zellweger. The simultaneous presence of these unique light microscopical characteristics may be of diagnostic value.

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Year:  1986        PMID: 2430454     DOI: 10.1002/ajmg.1320250210

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  25 in total

1.  X-linked recessive chondrodysplasia punctata with XY translocation in a stillborn fetus.

Authors:  L Van Maldergem; M Espeel; F Roels; C Petit; G Dacremont; R J Wanders; A Verloes; Y Gillerot
Journal:  Hum Genet       Date:  1991-10       Impact factor: 4.132

2.  Liver and chorion cytochemistry.

Authors:  F Roels; B De Prest; G De Pestel
Journal:  J Inherit Metab Dis       Date:  1995       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

4.  Phytanic acid alpha-oxidation and complementation analysis of classical Refsum and peroxisomal disorders.

Authors:  B T Poll-The; O H Skjeldal; O Stokke; A Poulos; F Demaugre; J M Saudubray
Journal:  Hum Genet       Date:  1989-01       Impact factor: 4.132

5.  Polarizing inclusions in some organs of children with congenital peroxisomal diseases (Zellweger's, Refsum's, chondrodysplasia punctata (rhizomelic form), X-linked adrenoleukodystrophy).

Authors:  I Kerckaert; K P Dingemans; H S Heymans; J Vamecq; F Roels
Journal:  J Inherit Metab Dis       Date:  1988       Impact factor: 4.982

6.  Peroxisomal integral membrane proteins in livers of patients with Zellweger syndrome, infantile Refsum's disease and X-linked adrenoleukodystrophy.

Authors:  G M Small; M J Santos; T Imanaka; A Poulos; D M Danks; H W Moser; P B Lazarow
Journal:  J Inherit Metab Dis       Date:  1988       Impact factor: 4.982

7.  Detection of peroxisomes in human liver and kidney fixed with formalin and embedded in paraffin: the use of catalase and lipid beta-oxidation enzymes as immunocytochemical markers.

Authors:  J A Litwin; A Völkl; J Stachura; H D Fahimi
Journal:  Histochem J       Date:  1988-03

8.  Establishment of a normal range of morphometric values for peroxisomes in paediatric liver.

Authors:  J L Hughes; A J Bourne; A Poulos
Journal:  Virchows Arch A Pathol Anat Histopathol       Date:  1993

9.  Peroxisomal fatty acid beta-oxidation in relation to the accumulation of very long chain fatty acids in cultured skin fibroblasts from patients with Zellweger syndrome and other peroxisomal disorders.

Authors:  R J Wanders; C W van Roermund; M J van Wijland; R B Schutgens; J Heikoop; H van den Bosch; A W Schram; J M Tager
Journal:  J Clin Invest       Date:  1987-12       Impact factor: 14.808

10.  Different types of peroxisomes in human duodenal epithelium.

Authors:  F Roels; M Espeel; M Pauwels; D De Craemer; H J Egberts; P van der Spek
Journal:  Gut       Date:  1991-08       Impact factor: 23.059

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