Literature DB >> 1705185

Glyceryl ethers in peroxisomal disease.

A Poulos1, A Bankier, K Beckman, D Johnson, E F Robertson, P Sharp, L Sheffield, H Singh, S Usher, G Wise.   

Abstract

1-O-Alkyl and 1-O-alk-1-enyl (plasmalogens) glyceryl ether lipid levels were measured in post-mortem brain and/or liver biopsies from 7 patients with ultrastructural and biochemical evidence of a defect in peroxisomal biogenesis and/or enzymological evidence of a disturbance in ether lipid synthesis. Near normal levels of both species of glyceryl ether lipids were found in neonatal adrenoleukodystrophy and infantile Refsum's disease but marked deficiencies were found in Zellweger's syndrome and rhizomelic chondrodysplasia punctata, the latter manifesting the most profound reduction in ether lipid levels. These observations suggest that little ether lipid biosynthesis occurs in vivo in rhizomelic chondrodysplasia punctata or Zellweger's syndrome. However, in some phenotypes with apparently gross reductions in peroxisomal numbers, e.g. neonatal adrenoleukodystrophy and infantile Refsom's disease, there is significant ether lipid synthesis in liver and brain.

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Year:  1991        PMID: 1705185     DOI: 10.1111/j.1399-0004.1991.tb02980.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  4 in total

1.  2-arachidonyl glyceryl ether, an endogenous agonist of the cannabinoid CB1 receptor.

Authors:  L Hanus; S Abu-Lafi; E Fride; A Breuer; Z Vogel; D E Shalev; I Kustanovich; R Mechoulam
Journal:  Proc Natl Acad Sci U S A       Date:  2001-03-20       Impact factor: 11.205

2.  Ultrastructure and immunocytochemistry of hepatic peroxisomes in rhizomelic chondrodysplasia punctata.

Authors:  J L Hughes; A Poulos; D I Crane; C W Chow; L J Sheffield; D Sillence
Journal:  Eur J Pediatr       Date:  1992-11       Impact factor: 3.183

3.  Plasmalogen deficiency in cerebral adrenoleukodystrophy and its modulation by lovastatin.

Authors:  Mushfiquddin Khan; Jaspreet Singh; Inderjit Singh
Journal:  J Neurochem       Date:  2008-06-07       Impact factor: 5.372

4.  Fatty acid synthesis from [2-14C]acetate in normal and peroxisome-deficient (Zellweger) fibroblasts.

Authors:  A Poulos; S Usher; B C Paton
Journal:  Lipids       Date:  1993-02       Impact factor: 1.880

  4 in total

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