Literature DB >> 2755305

Polyunsaturated fatty acid changes suggesting a new enzymatic defect in Zellweger syndrome.

M Martinez1.   

Abstract

The fatty acid composition of red blood cells, fibroblasts, forebrain, liver and kidney were studied in a 3-month-old infant who died from Zellweger Syndrome, and the results were compared with those of age-matched controls. Besides a typical increase in the very long chain fatty acids 26:0 and 26:1 and a great reduction in the plasmalogen levels, confirming the diagnosis of Zellweger Syndrome, some striking changes in the polyunsaturated fatty acid patterns were discovered. The most important was a very drastic decrease in the values of 22:6 omega 3 and 22:5 omega 6, the two products of delta 4-desaturation. In the kidney, the level of 22:6 omega 3 fell below that of 26:0. Consequently, the ratio 26:0/22:6 omega 3 (and 26:1/22:6 omega 3) was most useful in emphasizing the fatty acid anomalies, especially in renal tissue, where the 26:0/22:6 omega 3 ratio increased to almost 200 times the normal values. Other significant, although less consistent fatty acid alterations were increases in 18:2 omega 6, 18:3 omega 6, 20:3 omega 6, 18:4 omega 3 and 20:4 omega 3, and a decrease in 20:4 omega 6 in some tissues. The existence is proposed of a new enzyme defect in peroxisomal disorders, involving the desaturase system of long chain polyunsaturated fatty acids.

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Year:  1989        PMID: 2755305     DOI: 10.1007/BF02535160

Source DB:  PubMed          Journal:  Lipids        ISSN: 0024-4201            Impact factor:   1.880


  23 in total

1.  ALTERATION OF THE FATTY ACID COMPOSITION OF BRAIN LIPIDS BY VARYING LEVELS OF DIETARY ESSENTIAL FATTY ACIDS.

Authors:  H MOHRHAUER; R T HOLMAN
Journal:  J Neurochem       Date:  1963-07       Impact factor: 5.372

Review 2.  The cerebro-hepato-renal (Zellweger) syndrome and other peroxisomal disorders.

Authors:  H Zellweger
Journal:  Dev Med Child Neurol       Date:  1987-12       Impact factor: 5.449

3.  Cerebro-hepato-renal (Zellweger) syndrome and neonatal adrenoleukodystrophy: similarities in phenotype and accumulation of very long chain fatty acids.

Authors:  F R Brown; A J McAdams; J W Cummins; R Konkol; I Singh; A B Moser; H W Moser
Journal:  Johns Hopkins Med J       Date:  1982-12

4.  Polyunsaturated fatty acid lipidosis. II. Lipid biochemical studies.

Authors:  L Svennerholm; B Hagberg; M Haltia; P Sourander; M T Vanier
Journal:  Acta Paediatr Scand       Date:  1975-05

5.  Serial section analysis of mouse hepatic peroxisomes.

Authors:  K Gorgas
Journal:  Anat Embryol (Berl)       Date:  1985

6.  Modification of the fatty acid composition of cultured human fibroblasts.

Authors:  A A Spector; R E Kiser; G M Denning; S W Koh; L E DeBault
Journal:  J Lipid Res       Date:  1979-05       Impact factor: 5.922

7.  Lipids of the developing human retina: I. Total fatty acids, plasmalogens, and fatty acid composition of ethanolamine and choline phosphoglycerides.

Authors:  M Martinez; A Ballabriga; J J Gil-Gibernau
Journal:  J Neurosci Res       Date:  1988-08       Impact factor: 4.164

8.  Influence of environmental medium on fatty acid composition of human cells: leukocytes and fibroblasts.

Authors:  B Delplanque; B Jacotot
Journal:  Lipids       Date:  1987-04       Impact factor: 1.880

9.  Subcellular localization of acyl coenzyme A: dihydroxyacetone phosphate acyltransferase in rat liver peroxisomes (microbodies).

Authors:  A K Hajra; C L Burke; C L Jones
Journal:  J Biol Chem       Date:  1979-11-10       Impact factor: 5.157

10.  Ultrastructural and cytochemical demonstration of peroxisomes in cultured fibroblasts from patients with peroxisomal deficiency disorders.

Authors:  J A Arias; A B Moser; S L Goldfischer
Journal:  J Cell Biol       Date:  1985-05       Impact factor: 10.539

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  17 in total

1.  Dietary docosahexaenoic acid has little effect on peroxisomes in healthy mice.

Authors:  D De Craemer; M Pauwels; C Van den Branden
Journal:  Lipids       Date:  1996-11       Impact factor: 1.880

2.  Docosahexaenoic acid therapy in docosahexaenoic acid-deficient patients with disorders of peroxisomal biogenesis.

Authors:  M Martinez
Journal:  Lipids       Date:  1996-03       Impact factor: 1.880

Review 3.  Plasmalogens, phospholipase A2, and docosahexaenoic acid turnover in brain tissue.

Authors:  A A Farooqui; L A Horrocks
Journal:  J Mol Neurosci       Date:  2001 Apr-Jun       Impact factor: 3.444

Review 4.  Restoring the DHA levels in the brains of Zellweger patients.

Authors:  M Martinez
Journal:  J Mol Neurosci       Date:  2001 Apr-Jun       Impact factor: 3.444

5.  Deficiency of a Retinal Dystrophy Protein, Acyl-CoA Binding Domain-containing 5 (ACBD5), Impairs Peroxisomal β-Oxidation of Very-long-chain Fatty Acids.

Authors:  Yuichi Yagita; Kyoko Shinohara; Yuichi Abe; Keiko Nakagawa; Mohammed Al-Owain; Fowzan S Alkuraya; Yukio Fujiki
Journal:  J Biol Chem       Date:  2016-11-29       Impact factor: 5.157

6.  Hepatic microsomal and peroxisomal docosahexaenoate biosynthesis during piglet development.

Authors:  Z Li; M L Kaplan; D L Hachey
Journal:  Lipids       Date:  2000-12       Impact factor: 1.880

7.  Plasma and red blood cell fatty acids in peroxisomal disorders.

Authors:  A B Moser; D S Jones; G V Raymond; H W Moser
Journal:  Neurochem Res       Date:  1999-02       Impact factor: 3.996

8.  The Δ4-desaturation pathway for DHA biosynthesis is operative in the human species: differences between normal controls and children with the Zellweger syndrome.

Authors:  Manuela Martinez; Natalia Ichaso; Fernando Setien; Nuria Durany; Xiao Qiu; William Roesler
Journal:  Lipids Health Dis       Date:  2010-09-09       Impact factor: 3.876

9.  Blood polyunsaturated fatty acids in patients with peroxisomal disorders. A multicenter study.

Authors:  M Martinez; I Mougan; M Roig; A Ballabriga
Journal:  Lipids       Date:  1994-04       Impact factor: 1.880

10.  Abnormal lipid and fatty acid compositions of kidneys from mice with polycystic kidney disease.

Authors:  H M Aukema; T Yamaguchi; H Takahashi; B Celi; B J Holub
Journal:  Lipids       Date:  1992-06       Impact factor: 1.880

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