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亨特综合征:艾杜糖醛酸-2-硫酸酯酶cDNA克隆的分离及患者DNA分析

Hunter syndrome: isolation of an iduronate-2-sulfatase cDNA clone and analysis of patient DNA.

作者信息

Wilson P J, Morris C P, Anson D S, Occhiodoro T, Bielicki J, Clements P R, Hopwood J J

机构信息

Department of Chemical Pathology, Adelaide Children's Hospital, Australia.

出版信息

Proc Natl Acad Sci U S A. 1990 Nov;87(21):8531-5. doi: 10.1073/pnas.87.21.8531.

Abstract

Iduronate 2-sulfatase (IDS, EC 3.1.6.13) is required for the lysosomal degradation of heparan sulfate and dermatan sulfate. Mutations causing IDS deficiency in humans result in the lysosomal storage of these glycosaminoglycans and Hunter syndrome, an X chromosome-linked disease. We have isolated and sequenced a 2.3-kilobase cDNA clone coding for the entire sequence of human IDS. Analysis of the deduced 550-amino acid IDS precursor sequence indicates that IDS has a 25-amino acid amino-terminal signal sequence, followed by 8 amino acids that are removed from the proprotein. An internal proteolytic cleavage occurs to produce the mature IDS present in human liver shown to contain a 42-kDa polypeptide N-terminal to a 14-kDa polypeptide. The IDS sequence has strong sequence homology with other sulfatases (such as sea urchin arylsulfatase, human arylsulfatases A, B, and C, and human glucosamine 6-sulfatase), suggesting that the sulfatases comprise an evolutionarily related family of genes that arose by gene duplication and divergent evolution. The arylsulfatases have a greater homology with each other than with the non-arylsulfatases (IDS and glucosamine 6-sulfatase). The IDS cDNA detected RNA species of 5.7, 5.4, 2.1, and 1.4 kilobases in human placental RNA and revealed structural alterations and gross deletions of the IDS gene in many of the clinically severe Hunter syndrome patients studied.

摘要

艾杜糖醛酸2-硫酸酯酶(IDS,EC 3.1.6.13)是硫酸乙酰肝素和硫酸皮肤素溶酶体降解所必需的。导致人类IDS缺乏的突变会导致这些糖胺聚糖在溶酶体中蓄积,并引发亨特综合征,这是一种X染色体连锁疾病。我们分离并测序了一个2.3千碱基的cDNA克隆,其编码人类IDS的完整序列。对推导的550个氨基酸的IDS前体序列分析表明,IDS有一个25个氨基酸的氨基末端信号序列,随后是从原蛋白中去除的8个氨基酸。发生内部蛋白水解切割以产生存在于人类肝脏中的成熟IDS,显示其包含一个42 kDa多肽,位于一个14 kDa多肽的N末端。IDS序列与其他硫酸酯酶(如海胆芳基硫酸酯酶、人类芳基硫酸酯酶A、B和C以及人类氨基葡萄糖6-硫酸酯酶)有很强的序列同源性,这表明硫酸酯酶构成了一个通过基因复制和趋异进化产生的进化相关基因家族。芳基硫酸酯酶之间的同源性比与非芳基硫酸酯酶(IDS和氨基葡萄糖6-硫酸酯酶)之间的同源性更高。IDS cDNA在人胎盘RNA中检测到5.7、5.4、2.1和1.4千碱基的RNA种类,并在许多研究的临床严重亨特综合征患者中揭示了IDS基因的结构改变和大片段缺失。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9ba5/54990/6abfdbb00b33/pnas01046-0356-a.jpg

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