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多种硫酸酯酶缺乏症:催化无活性的硫酸酯酶由逆转录病毒导入的硫酸酯酶cDNA表达。

Multiple sulfatase deficiency: catalytically inactive sulfatases are expressed from retrovirally introduced sulfatase cDNAs.

作者信息

Rommerskirch W, von Figura K

机构信息

Georg-August-Universität, Abteilung Biochemie II, Göttingen, Federal Republic of Germany.

出版信息

Proc Natl Acad Sci U S A. 1992 Apr 1;89(7):2561-5. doi: 10.1073/pnas.89.7.2561.

Abstract

Multiple sulfatase deficiency (MSD) is an inherited lysosomal storage disease characterized by the deficiency of at least seven sulfatases. The basic defect in MSD is thought to be in a post-translational modification common to all sulfatases. In accordance with this concept, RNAs of normal size and amount were detected in MSD fibroblasts for three sulfatases tested. cDNAs encoding arylsulfatase A, arylsulfatase B, or steroid sulfatase were introduced into MSD fibroblasts and fibroblasts with a single sulfatase deficiency by retroviral gene transfer. Infected fibroblasts overexpressed the respective sulfatase polypeptides. While in single-sulfatase-deficiency fibroblasts a concomitant increase of sulfatase activities was observed, MSD fibroblasts expressed sulfatase polypeptides with a severely diminished catalytic activity. From these results we conclude that the mutation in MSD severely decreases the capacity of a co- or post-translational process that renders sulfatases enzymatically active or prevents their premature inactivation.

摘要

多种硫酸酯酶缺乏症(MSD)是一种遗传性溶酶体贮积病,其特征是至少七种硫酸酯酶缺乏。MSD的基本缺陷被认为存在于所有硫酸酯酶共有的翻译后修饰中。根据这一概念,在检测的三种硫酸酯酶的MSD成纤维细胞中检测到了正常大小和数量的RNA。通过逆转录病毒基因转移,将编码芳基硫酸酯酶A、芳基硫酸酯酶B或类固醇硫酸酯酶的cDNA导入MSD成纤维细胞和单硫酸酯酶缺乏的成纤维细胞中。被感染的成纤维细胞过表达各自的硫酸酯酶多肽。虽然在单硫酸酯酶缺乏的成纤维细胞中观察到硫酸酯酶活性随之增加,但MSD成纤维细胞表达的硫酸酯酶多肽催化活性严重降低。从这些结果我们得出结论,MSD中的突变严重降低了使硫酸酯酶具有酶活性或防止其过早失活的共翻译或翻译后过程的能力。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/971b/48701/ef6fb67d23d3/pnas01081-0063-a.jpg

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