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溶酶体贮积症的产前诊断

Prenatal diagnosis of lysosomal storage diseases.

作者信息

Lake B D, Young E P, Winchester B G

机构信息

Department of Histopathology, Great Ormond Street Hospital for Children, London, UK.

出版信息

Brain Pathol. 1998 Jan;8(1):133-49. doi: 10.1111/j.1750-3639.1998.tb00141.x.

Abstract

The prenatal diagnosis of lysosomal storage disorders can be achieved, once the diagnosis is confirmed in the index case, by a variety of techniques including analysis of amniotic fluid, asay of enzymic activity in cultured amniotic fluid cells, cultured chorionic villus cells and by direct assay of activity in chorionic villus samples. These studies can be accompanied by ultrastructural observations which give an independent means of diagnosis. In some instances molecular genetic studies for mutation detection or linkage analysis are appropriate for prenatal diagnosis. Pseudodeficiencies of some of the lysosomal enzymes, which cause no clinical problems, can complicate the initial diagnosis particularly in metachromatic leucodystrophy where the pseudodeficiency is more common than the disease itself. Mutation analysis as well as enzyme assay is necessary not only in the index case but also in the parents before the same techniques are applied to a sample for prenatal diagnosis. A large number of lysosomal storage disorders may present as fetal hydrops and the diagnosis can be established at this late stage by fetal blood sampling and examination by microscopy as well as by biochemical assay of the appropriate enzyme or metabolite in amniotic fluid. All prenatal diagnoses in which an affected fetus is indicated should have confirmation of the diagnosis as soon as possible to reassure anxious parents, and to act as audit of the laboratory's competence to undertake prenatal diagnosis. A combined approach to prenatal diagnosis involving biochemical, molecular genetic and morphological studies is recommended.

摘要

一旦在先证者中确诊溶酶体贮积症,便可通过多种技术进行产前诊断,这些技术包括羊水分析、培养的羊水细胞、培养的绒毛膜绒毛细胞中的酶活性测定,以及绒毛膜绒毛样本中的活性直接测定。这些研究可辅以超微结构观察,提供独立的诊断方法。在某些情况下,用于突变检测或连锁分析的分子遗传学研究适用于产前诊断。一些溶酶体酶的假缺陷不会引起临床问题,但可能使初始诊断复杂化,尤其是在异染性脑白质营养不良中,假缺陷比疾病本身更常见。不仅在先证者中,而且在父母中进行突变分析和酶测定都是必要的,然后才能将相同技术应用于产前诊断样本。大量溶酶体贮积症可能表现为胎儿水肿,在这个晚期阶段,可通过采集胎儿血液并进行显微镜检查,以及对羊水中适当的酶或代谢物进行生化测定来确诊。所有提示有患病胎儿的产前诊断都应尽快确诊,以安抚焦虑的父母,并作为对实验室进行产前诊断能力的审核。建议采用生化、分子遗传学和形态学研究相结合的方法进行产前诊断。

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Prenatal diagnosis of lysosomal storage diseases.溶酶体贮积症的产前诊断
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