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PMID: 2430454 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

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

American journal of medical genetics ·Vol. 25 ·No. 2 ·1986-10-00 ·Pages 257-71

Roels F, Cornelis A, Poll-The BT, Aubourg P, Ogier H, Scotto J, Saudubray JM

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.

MeSH Terms
Birefringence Catalase/metabolism Child Child, Preschool Female Histocytochemistry Humans Liver/enzymology,ultrastructure Macrophages/ultrastructure Male Microbodies/enzymology,ultrastructure Microscopy, Electron Refsum Disease/enzymology,etiology,pathology
Chemicals
Catalase
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Roels F
Cornelis A
Poll-The B T
Aubourg P
Ogier H
Scotto J
Saudubray J M
Article Info
Journal
American journal of medical genetics
Abbr.
Am J Med Genet
ISSN
0148-7299
Published
1986-10-00
Pages
257-71
Language
English
Region
United States
NLM ID
7708900
Subset
IM
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