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第8外显子缺失导致一名非裔美国天冬氨酰葡糖胺尿症(AGU)患者出现糖基天冬酰胺酶缺乏。

Deletion of exon 8 causes glycosylasparaginase deficiency in an African American aspartylglucosaminuria (AGU) patient.

作者信息

Fisher K J, Aronson N N

机构信息

Department of Molecular and Cell Biology, Althouse Lab, Pennsylvania State University, University Park 16802.

出版信息

FEBS Lett. 1991 Aug 19;288(1-2):173-8. doi: 10.1016/0014-5793(91)81028-7.

Abstract

We have indentified a GT-to-TT transversion at the splice donor site of intron 8 in the glycosylasparaginase gene from an African American aspartylglucosaminuria (AGU) patient. This mutation causes abnormal splicing of glycosylasparaginase pre-mRNA by joining exon 7 to 9 and excluding 134 bp exon 8. The effect of the mutation is compounded by a frame shift that occurs after the deletion site resulting in premature translational termination. The truncated AGU protein was neither catalytically active nor processed into mature alpha and beta subunits. Both this and a previously characterized Finnish AGU mutation appear to affect folding of the single-chain precursor of glycosylasparaginase and thereby prevent transport of the enzyme to lysosomes.

摘要

我们在一名非裔美国天冬氨酰葡糖胺尿症(AGU)患者的氨基己糖苷酶基因第8内含子的剪接供体位点发现了一个从GT到TT的颠换。该突变通过将外显子7与外显子9连接并排除134 bp的外显子8,导致氨基己糖苷酶前体mRNA的异常剪接。该突变的影响因缺失位点后发生的移码而加剧,导致过早的翻译终止。截短的AGU蛋白既无催化活性,也未加工成成熟的α和β亚基。这个突变以及之前鉴定的一个芬兰AGU突变似乎都影响了氨基己糖苷酶单链前体的折叠,从而阻止了该酶向溶酶体的转运。

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