Brooks D A, McCourt P A, Gibson G J, Hopwood J J
Department of Chemical Pathology, Adelaide Childrens Hospital, Australia.
J Inherit Metab Dis. 1990;13(1):108-20. doi: 10.1007/BF01799338.
The low abundance lysosomal enzyme N-acetylgalactosamine 4-sulphatase (4-sulphatase) has been quantified using a microimmunopurification step and a monoclonal-based ELISA detection system. The assay is similar in principle to a two-site ELISA but uses a single monoclonal antibody against one epitope to bind 4-sulphatase in two separate assay steps. The sensitivity of this assay is sufficient to allow the quantification of 4-sulphatase in human cultured skin fibroblasts derived from normal controls and patients deficient in 4-sulphatase activity (mucopolysaccharidosis type VI or Maroteaux-Lamy syndrome). The results obtained suggest a range of mucopolysaccharidosis type VI or 4-sulphatase deficient mutants, from those expressing little or no quantifiable 4-sulphatase protein to those examples with quantifiable levels of 4-sulphatase protein which is enzymically inactive. Phenotypic variability in patients with a 4-sulphatase deficiency may therefore be partially attributed to a range of protein expressions. The method should allow the determination of 4-sulphatase specific activity in mucopolysaccharidosis type VI patients.
已使用微免疫纯化步骤和基于单克隆抗体的酶联免疫吸附测定(ELISA)检测系统对低丰度溶酶体酶N-乙酰半乳糖胺4-硫酸酯酶(4-硫酸酯酶)进行了定量。该测定法在原理上与双位点ELISA相似,但在两个独立的测定步骤中使用针对一个表位的单克隆抗体来结合4-硫酸酯酶。该测定法的灵敏度足以对来自正常对照和4-硫酸酯酶活性缺乏患者(VI型粘多糖贮积症或马罗泰克斯-拉米综合征)的人培养皮肤成纤维细胞中的4-硫酸酯酶进行定量。所获得的结果表明,VI型粘多糖贮积症或4-硫酸酯酶缺陷突变体的范围很广,从那些几乎不表达或无法定量的4-硫酸酯酶蛋白的突变体到那些具有可定量水平但酶无活性的4-硫酸酯酶蛋白的例子。因此,4-硫酸酯酶缺乏患者的表型变异性可能部分归因于一系列蛋白质表达。该方法应能测定VI型粘多糖贮积症患者中4-硫酸酯酶的比活性。