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PMID: 7692128 Published · ppublish English Journal Article Research Support, Non-U.S. Gov't

The deficient degradation of synthetic 2- and 3-methyl-branched fatty acids in fibroblasts from patients with peroxisomal disorders.

Journal of inherited metabolic disease ·Vol. 16 ·No. 2 ·1993-00-00 ·Pages 381-91

Van Veldhoven PP, Huang S, Eyssen HJ, Mannaerts GP

Abstract

The oxidation of pristanic and phytanic acids by human skin fibroblasts was compared to that of their synthetic analogues, 2-methylpalmitic and 3-methylmargaric acids. The synthetic compounds and natural substrates were degraded at comparable rates in control and X-linked adrenoleukodystrophy fibroblasts. The alpha-decarboxylation of 3-methylmargaric acid, similarly to that of phytanic acid, was affected in Refsum disease and Zellweger syndrome, but not in X-linked adrenoleukodystrophy. The beta-oxidation of 2-methylpalmitic acid, similarly to that of pristanic acid, was deficient in fibroblasts derived from patients suffering from Zellweger syndrome, confirming the importance of peroxisomes in the breakdown of 2-methyl-branched fatty acids. No deficiency was observed in fibroblasts from X-linked adrenoleukodystrophy patients. The 1-14C-labelled 2- and 3-methyl-branched fatty acids, which are easier to synthesize that the natural analogues, are therefore valuable tools for the diagnosis of human peroxisomal disorders.

MeSH Terms
Adrenoleukodystrophy/genetics,metabolism Cell Line Fatty Acids/chemistry,metabolism Fibroblasts/metabolism Humans Kinetics Microbodies/metabolism Oxidation-Reduction Palmitates/metabolism Phytanic Acid/metabolism Refsum Disease/metabolism X Chromosome Zellweger Syndrome/metabolism
Chemicals
Fatty Acids Palmitates Phytanic Acid 3-methylheptadecanoic acid pristanic acid methyl palmitate
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Van Veldhoven P P
Katolieke Universiteit Leuven, Afdeling Farmakologie, Belgium.
Huang S
Eyssen H J
Mannaerts G P
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Article Info
Journal
Journal of inherited metabolic disease
Abbr.
J Inherit Metab Dis
ISSN
0141-8955
Published
1993-00-00
Pages
381-91
Language
English
Region
United States
NLM ID
7910918
Subset
IM
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