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Altered phospholipid molecular species and glycolipid composition in brain, liver and fibroblasts of Zellweger syndrome.

Abstract：

:We studied the altered molecular species of lipids in brain and liver tissues, and fibroblasts from patients with Zellweger syndrome (ZS). ZS cerebellum samples contained a higher amount of sphingomyelin with shorter chain fatty acids compared to that in normal controls. The amount of phosphatidylethanolamine (PE) was less than half of that in controls, with the absence of the PE-type of plasmalogen. Gangliosides were accumulated in the brains and fibroblasts of ZS patients. To investigate whether or not impaired beta-oxidation of very long chain fatty acids and/or plasmalogen synthesis affects glycolipids metabolism, RNAi of peroxisomal acylCo-A oxidase (ACOX1) and glyceronephosphate O-acyltransferase (GNPAT) was performed using cultured neural cells. In neuronal F3-Ngn1 cells, ACOX1 and GNPAT silencing up-regulated ceramide galactosyltransferase (UGT8) mRNA expression, and down-regulated UDP-glucose ceramide glucosyltransferase (UGCG). These results suggest that both impaired beta-oxidation of very long chain fatty acids and plasmalogen synthesis affect glycolipid metabolism in neuronal cells.

journal_name

Neurosci Lett

journal_title

Neuroscience letters

authors

Miyazaki C,Saitoh M,Itoh M,Yamashita S,Miyagishi M,Takashima S,Moser AB,Iwamori M,Mizuguchi M

doi

10.1016/j.neulet.2013.07.045

subject

Has Abstract

pub_date

2013-09-27 00:00:00

pages

71-5

eissn

0304-3940

issn

1872-7972

pii

S0304-3940(13)00707-6

journal_volume

552

pub_type

杂志文章

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