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β-葡萄糖醛酸酶缺乏症:对一个患病家庭的酶学研究及产前诊断

Beta-glucuronidase deficiency: enzyme studies in an affected family and prenatal diagnosis.

作者信息

Maire I, Mandon G, Zabot M T, Mathieu M, Guibaud P

出版信息

J Inherit Metab Dis. 1980;2(2):29-34. doi: 10.1007/BF01799071.

Abstract

A beta-glucuronidase deficiency found in serum, leukocytes and fibroblasts and an increased [35S]sulphate incorporation in fibroblasts led us to diagnose two cases of type VII mucopolysaccharidosis in one family. In spite of the wide distribution of activities in serum from controls, decreased beta-glucuronidase activity allowed us to demonstrate the heterozygous status of the parents and two other children. Following these studies, and antenatal diagnosis was performed when the mother was pregnant again; amniotic fluid and cultured amniotic cells were used for enzyme activity determination. A heterozygous fetus was suspected and confirmed after birth. The reliability of various biological materials for enzymatic diagnosis and existence of genetic variants in the normal population are discussed.

摘要

在血清、白细胞和成纤维细胞中发现β-葡萄糖醛酸酶缺乏,且成纤维细胞中[35S]硫酸盐掺入增加,这使我们在一个家族中诊断出两例VII型黏多糖贮积症。尽管对照血清中的活性分布广泛,但β-葡萄糖醛酸酶活性降低使我们能够证实父母和另外两个孩子的杂合状态。在这些研究之后,母亲再次怀孕时进行了产前诊断;羊水和培养的羊膜细胞用于酶活性测定。怀疑胎儿为杂合子,并在出生后得到证实。讨论了各种生物材料用于酶诊断的可靠性以及正常人群中基因变异的存在情况。

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