Literature DB >> 13129591

Gas chromatography/mass spectrometry analysis of very long chain fatty acids, docosahexaenoic acid, phytanic acid and plasmalogen for the screening of peroxisomal disorders.

Yasuhiko Takemoto1, Yasuyuki Suzuki, Ryoko Horibe, Nobuyuki Shimozawa, Ronald J A Wanders, Naomi Kondo.   

Abstract

Very long chain fatty acids (VLCFAs) and docosahexaenoic acid (DHA), phytanic acid, and plasmalogens are usually measured individually. A novel method for the screening of peroxisomal disorders, using gas chromatography/mass spectrometry (GC/MS), was developed. Saturated and unsaturated fatty acids, including VLCFAs and DHA, phytanic acid, and plasmalogen were detected by a selected ion monitoring-electron impact method, using 100 microl of serum or plasma. Methyl-esterification and extraction could be done in one tube, and data were obtained within 4 h. All patients with Zellweger syndrome (ZS), X-linked adrenoleukodystrophy (ALD), isolated deficiency of peroxisomal beta-oxidation enzyme, and most ALD carriers showed increased VLCFA ratios, including C24:0/C22:0, C25:0/C22:0 and C26:0/C22:0. The ratio of DHA to palmitic acid (C16:0) and plasmalogen (measured as hexadecanal dimethyl acetal) to C16:0 in ZS patients was significantly lower than for the controls (P<0.001 for healthy high school students, P<0.05 for infants with other disorders). Plasmalogen was also decreased in patients with isolated deficiency of plasmalogen biosynthesis. Two of eight patients with ZS, two of four with RCDP, and all of three classical Refsum patients showed increased levels of phytanic acid. This method will simplify the screening for peroxisomal disorders.

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Year:  2003        PMID: 13129591     DOI: 10.1016/s0387-7604(03)00033-0

Source DB:  PubMed          Journal:  Brain Dev        ISSN: 0387-7604            Impact factor:   1.961


  23 in total

Review 1.  Peroxisome biogenesis disorders in the Zellweger spectrum: An overview of current diagnosis, clinical manifestations, and treatment guidelines.

Authors:  Nancy E Braverman; Gerald V Raymond; William B Rizzo; Ann B Moser; Mark E Wilkinson; Edwin M Stone; Steven J Steinberg; Michael F Wangler; Eric T Rush; Joseph G Hacia; Mousumi Bose
Journal:  Mol Genet Metab       Date:  2015-12-23       Impact factor: 4.797

2.  A Novel Double Mutation in the ABCD1 Gene in a Patient with X-linked Adrenoleukodystrophy: Analysis of the Stability and Function of the Mutant ABCD1 Protein.

Authors:  Masashi Morita; Junpei Kobayashi; Kozue Yamazaki; Kosuke Kawaguchi; Ayako Honda; Kenji Sugai; Nobuyuki Shimozawa; Reiji Koide; Tsuneo Imanaka
Journal:  JIMD Rep       Date:  2013-02-12

Review 3.  Measuring brain lipids.

Authors:  Glyn Dawson
Journal:  Biochim Biophys Acta       Date:  2015-02-18

Review 4.  Meibomian glands, meibum, and meibogenesis.

Authors:  Igor A Butovich
Journal:  Exp Eye Res       Date:  2017-06-29       Impact factor: 3.467

5.  Mild Zellweger syndrome due to a novel PEX6 mutation: correlation between clinical phenotype and in silico prediction of variant pathogenicity.

Authors:  Małgorzata Rydzanicz; Teresa Joanna Stradomska; Elżbieta Jurkiewicz; Ewa Jamroz; Piotr Gasperowicz; Grażyna Kostrzewa; Rafał Płoski; Anna Tylki-Szymańska
Journal:  J Appl Genet       Date:  2017-10-18       Impact factor: 3.240

6.  Dual effect of docosahexaenoic acid (attenuation or amplification) on C22:0-, C24:0-, and C26:0-induced mitochondrial dysfunctions and oxidative stress on human neuronal SK-N-BE cells.

Authors:  A Zarrouk; T Nury; J M Riedinger; O Rouaud; M Hammami; G Lizard
Journal:  J Nutr Health Aging       Date:  2015-02       Impact factor: 4.075

7.  Effect of Lorenzo's Oil on Hepatic Gene Expression and the Serum Fatty Acid Level in abcd1-Deficient Mice.

Authors:  Masashi Morita; Ayako Honda; Akira Kobayashi; Yuichi Watanabe; Shiro Watanabe; Kosuke Kawaguchi; Shigeo Takashima; Nobuyuki Shimozawa; Tsuneo Imanaka
Journal:  JIMD Rep       Date:  2017-05-31

8.  Detection and Quantification of Free Radicals in Peroxisomal Disorders: A Comparative Study with Oxidative Stress Parameters.

Authors:  Sohair Abd-El Mawgood Abd-Elmaksoud; Hala El-Bassyouni; Hanan Afifi; Manal Micheal Thomas; Alshaymaa Ahmed Ibrahim; Aliaa Shalaby; Tamer Ahmed Abdel Hamid; Nehal Abdel Hamid; Hany El-Ghobary
Journal:  J Clin Diagn Res       Date:  2015-11-01

9.  Direct microwave transesterification of fingertip prick blood samples for fatty acid determinations.

Authors:  Jeff M Armstrong; Adam H Metherel; Ken D Stark
Journal:  Lipids       Date:  2007-12-15       Impact factor: 1.880

10.  Laboratory diagnosis of disorders of peroxisomal biogenesis and function: a technical standard of the American College of Medical Genetics and Genomics (ACMG).

Authors:  Irene De Biase; Silvia Tortorelli; Lisa Kratz; Steven J Steinberg; Kristina Cusmano-Ozog; Nancy Braverman
Journal:  Genet Med       Date:  2019-12-11       Impact factor: 8.822

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