Takemoto Yasuhiko, Suzuki Yasuyuki, Horibe Ryoko, Shimozawa Nobuyuki, Wanders Ronald J A, Kondo Naomi
Department of Pediatrics, Gifu University School of Medicine, Tsukasa-machi 40, Gifu 500-8705, Japan.
Brain Dev. 2003 Oct;25(7):481-7. doi: 10.1016/s0387-7604(03)00033-0.
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.
极长链脂肪酸(VLCFAs)、二十二碳六烯酸(DHA)、植烷酸和缩醛磷脂通常分别进行检测。开发了一种使用气相色谱/质谱法(GC/MS)筛查过氧化物酶体疾病的新方法。通过选择离子监测-电子轰击法,使用100微升血清或血浆检测饱和脂肪酸和不饱和脂肪酸,包括VLCFAs、DHA、植烷酸和缩醛磷脂。甲基酯化和提取可在同一试管中完成,4小时内即可获得数据。所有患有泽尔韦格综合征(ZS)、X连锁肾上腺脑白质营养不良(ALD)、过氧化物酶体β氧化酶孤立缺乏症的患者以及大多数ALD携带者的VLCFA比值均升高,包括C24:0/C22:0、C25:0/C22:0和C26:0/C22:0。ZS患者中DHA与棕榈酸(C16:0)的比值以及缩醛磷脂(以十六醛二甲基缩醛测量)与C16:0的比值显著低于对照组(健康高中生P<0.001,其他疾病婴儿P<0.05)。在缩醛磷脂生物合成孤立缺乏症患者中,缩醛磷脂也减少。8例ZS患者中有2例、4例RCDP患者中有2例以及所有3例经典型Refsum病患者的植烷酸水平升高。该方法将简化过氧化物酶体疾病的筛查。