Kolodny E H, Mumford R A
Adv Exp Med Biol. 1976;68:239-51. doi: 10.1007/978-1-4684-7735-1_16.
Metachromatic leukodystrophy and Maroteaux-Lamy syndrome can be diagnosed by assay of leukocyte or fibroblast arylsulfatase A and B activity with the fluorogenic substrate 4-methylumbelliferyl sulfate. The arylsulfatases are extracted into a 27000 x g supernatant by sonication in 0.9% sodium chloride and then separated with CM-32 on columns or in test tubes. In 0.05 M sodium acetate pH 6.0, arylsulfatase A is not absorbed while arylsulfatase B is retained by the resin. The arylsulfatase B is then eluted from the resin with 0.3 M sodium chloride. The arylsulfatase A activity obtained from normal leukocytes and fibroblasts is linear for the initial 10 minutes of the reaction, is stimulated 3-fold by 6 mM lead acetate and inhibited 80% by 0.24 mM silver nitrate. After separation with CM-32, the arylsulfatase B activity is stimulated 3-fold by Triton X-100 (0.1%). Arylsulfatase A but not arylsulfatase B is destroyed by heat (60 degrees). Both leukocyte and fibroblast arylsulfatase A activity was reduced to 11% of control values in metachromatic leukodystrophy. Essentially no arylsulfatase B activity was detected in cells from patients with Maroteaux-Lamy syndrome. Metachromatic leukodystrophy heterozygotes but not Maroteaux-Lamy syndrome heterozygotes can also be distinguished by this method. A heat inactivation technique utilizing the differential thermal stabilities of the two enzymes for diagnosis of patients with Marotezux-Lamy syndrome is also described. The advantages of these 4-methylumbelliferyl sulfate assay procedures over the p-nitrocatechol sulfate method of assay are greater sensitivity, selectivity for the desired enzyme and potential for use in large scale testing.
通过使用荧光底物4-甲基伞形酮基硫酸酯检测白细胞或成纤维细胞芳基硫酸酯酶A和B的活性,可诊断异染性脑白质营养不良和马罗泰克斯-拉米综合征。通过在0.9%氯化钠中超声处理,将芳基硫酸酯酶提取到27000×g的上清液中,然后在柱上或试管中用CM-32进行分离。在0.05M醋酸钠pH6.0条件下,芳基硫酸酯酶A不被吸附,而芳基硫酸酯酶B被树脂保留。然后用0.3M氯化钠从树脂上洗脱芳基硫酸酯酶B。从正常白细胞和成纤维细胞获得的芳基硫酸酯酶A活性在反应的最初10分钟呈线性,6mM醋酸铅可使其活性提高3倍,0.24mM硝酸银可使其活性抑制80%。用CM-32分离后,0.1%的 Triton X-100可使芳基硫酸酯酶B活性提高3倍。芳基硫酸酯酶A可被加热(60℃)破坏,而芳基硫酸酯酶B则不会。在异染性脑白质营养不良中,白细胞和成纤维细胞的芳基硫酸酯酶A活性均降至对照值的11%。在马罗泰克斯-拉米综合征患者的细胞中基本未检测到芳基硫酸酯酶B活性。通过这种方法也可区分异染性脑白质营养不良杂合子,但不能区分马罗泰克斯-拉米综合征杂合子。还描述了一种利用两种酶的不同热稳定性进行热失活技术,用于诊断马罗泰克斯-拉米综合征患者。与对硝基儿茶酚硫酸酯检测方法相比,这些4-甲基伞形酮基硫酸酯检测方法的优点是灵敏度更高、对所需酶的选择性更强以及具有大规模检测的潜力。