Okada S, Kato T, Miura S, Yabuuchi H, Nishigaki M, Kobata A, Chiyo H, Furuyama J I
Clin Chim Acta. 1978 Jun;86(2):159-67. doi: 10.1016/0009-8981(78)90129-8.
A method is described for the detection of abnormal oligosaccharides in a small (5 ml) volume of urine, employing filtration on a Bio Gel P-6 column, determination of neutral sugar and bound sialic acid, and determination of creatinine content. With this method increased urinary excretion of sialic acid-rich oligosaccharides has been detected in nine patients with mucolipidoses (five cases of mucolipidosis II and four patients of mucolipidosis, with beta-galactosidase deficiency). The filtration patterns of oligosaccharides in mucolipidoses were clearly distinguishable from those in other inborn errors of metabolism. Total excreted oligosaccharides were increased 5--30-fold in these patients; mucolipidosis II, 640--1350 microgram neutral sugar/mg creatinine; control 54 +/- 20 microgram neutral sugar/mg creatinine. The oligosaccharides consisted of three sialic acid-rich fractions and were common in both types of mucolipidosis. Our data indicate that hypersialyoligosacchariduria is the main biochemical feature of both types of mucolipidosis.
本文描述了一种检测少量(5毫升)尿液中异常寡糖的方法,该方法采用在生物凝胶P - 6柱上过滤、测定中性糖和结合唾液酸以及测定肌酐含量。通过这种方法,在9例粘脂贮积症患者(5例粘脂贮积症II型和4例β - 半乳糖苷酶缺乏的粘脂贮积症患者)中检测到富含唾液酸的寡糖尿排泄增加。粘脂贮积症中寡糖的过滤模式与其他先天性代谢缺陷中的明显不同。这些患者中总排泄寡糖增加了5 - 30倍;粘脂贮积症II型,640 - 1350微克中性糖/毫克肌酐;对照组为54±20微克中性糖/毫克肌酐。这些寡糖由三个富含唾液酸的部分组成,在两种类型的粘脂贮积症中都很常见。我们的数据表明,高唾液酸化寡糖尿症是两种类型粘脂贮积症的主要生化特征。