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[常染色体隐性多囊肾病的产前诊断]

[Prenatal diagnosis of autosomal recessive polycystic kidney disease].

作者信息

Gagnadoux M F, Attié T, Amiel J, Gigarel N, Bonnefont J P, Munnich A, Gubler M C, Antignac C

机构信息

Service de néphrologie pédiatrique, hôpital Necker-Enfants-malades, Paris, France.

出版信息

Arch Pediatr. 2000 Sep;7(9):942-7. doi: 10.1016/s0929-693x(00)90007-1.

Abstract

OBJECTIVE

Prenatal ultrasonographic detection of autosomal recessive polycystic kidney disease (ARPKD) is of poor reliability, especially in early pregnancy. Molecular genetics allows earlier diagnosis, from 11 weeks of gestation; however, since only indirect diagnosis is possible--the ARPKD gene being localized on chromosome 6 but not identified--the feasibility of molecular diagnosis requires several conditions: definitive diagnosis in the index case, availability of index case and parents' DNA, genetic informativity of the family at the ARPKD locus. Results and limits of this method are analyzed, using a series of 56 requests for prenatal diagnosis.

RESULTS

In eight of the 56 families ARPKD was excluded on the basis of histological (seven cases) and/or genetic (two cases) criteria. Molecular study was impossible in three families due to the lack of index case's DNA, and two other families were non-informative. Among the 43 families in which prenatal diagnosis was feasible, analysis of the haplotype of 35 fetuses issued from 29 families showed that 11 fetuses with the same haplotypes as that of the index case were affected, while 24 were not. No false positive or false negative result was reported.

CONCLUSIONS

Early and reliable prenatal diagnosis of recessive polycystic kidney disease is possible in nearly 80% of affected families.

摘要

目的

产前超声检查对常染色体隐性多囊肾病(ARPKD)的诊断可靠性较差,尤其是在孕早期。分子遗传学可在妊娠11周起进行更早的诊断;然而,由于只能进行间接诊断——ARPKD基因位于6号染色体上但尚未确定——分子诊断的可行性需要几个条件:先证者确诊、先证者及其父母的DNA可用、家族在ARPKD位点的遗传信息性。使用一系列56例产前诊断请求分析了该方法的结果和局限性。

结果

在56个家庭中的8个家庭中,根据组织学标准(7例)和/或遗传学标准(2例)排除了ARPKD。由于缺乏先证者的DNA,3个家庭无法进行分子研究,另外2个家庭无信息价值。在43个可行产前诊断的家庭中,对来自29个家庭的35例胎儿的单倍型分析表明,11例与先证者具有相同单倍型的胎儿患病,而24例未患病。未报告假阳性或假阴性结果。

结论

在近80%的患病家庭中,可进行早期且可靠的隐性多囊肾病产前诊断。

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