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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