Groebe H, Krins M, Schmidberger H, von Figura K, Harzer K, Kresse H, Paschke E, Sewell A, Ullrich K
Am J Hum Genet. 1980 Mar;32(2):258-72.
Two male patients, aged 6 and 25, both with normal intelligence and absence of neurological abnormalities, exhibited dysostosis multiplex, dwarfism, odontoid anomalies, cloudy corneas, exessive excretion of keratan sulfate, and abnormal urinary oligosaccharides. Leukocytes and fibroblasts of both patients were deficient in acid beta-galactosidase (beta-gal) and normal in N-acetylgalactosamine-6-sulfate sulfatase, the deficient enzyme in classical Morquio syndrome. The beta-gal deficiency was not due to an endogenous inhibitor, and the parents exhibited intermediate activities. Deficient beta-gal activity was observed toward p-nitrophenyl-beta-galactoside, 4-methylumbelliferyl-beta-galactoside (4 MU-beta-gal), lactose, GM1 ganglioside, keratan sulfate, and asialofetuin (ASF). Under standard assay conditions, the residual activity was similar for all substrates tested. Toward p-nitrophenyl-beta-glactoside, the mutant enzyme behaved as a Km variant.
两名男性患者,年龄分别为6岁和25岁,智力均正常且无神经学异常,表现出多发性骨发育异常、侏儒症、齿状突异常、角膜混浊、硫酸角质素排泄过多以及异常尿寡糖。两名患者的白细胞和成纤维细胞中酸性β-半乳糖苷酶(β-gal)缺乏,而N-乙酰半乳糖胺-6-硫酸硫酸酯酶正常,后者是典型Morquio综合征中的缺陷酶。β-gal缺乏并非由内源性抑制剂引起,其父母表现出中间活性。观察到对对硝基苯基-β-半乳糖苷、4-甲基伞形酮基-β-半乳糖苷(4-MU-β-gal)、乳糖、GM1神经节苷脂、硫酸角质素和去唾液酸胎球蛋白(ASF)的β-gal活性缺乏。在标准测定条件下,所有测试底物的残余活性相似。对于对硝基苯基-β-葡萄糖苷,突变酶表现为Km变体。