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产前诊断孤立性重度双侧脑室扩张胎儿的结局:系统评价和荟萃分析。

Outcome of fetuses with prenatal diagnosis of isolated severe bilateral ventriculomegaly: systematic review and meta-analysis.

机构信息

Fetal Medicine Unit, St George's University Hospital NHS Foundation Trust, London, UK.

出版信息

Ultrasound Obstet Gynecol. 2018 Aug;52(2):165-173. doi: 10.1002/uog.19038.

DOI:10.1002/uog.19038
PMID:29484752
Abstract

OBJECTIVE

To quantify from the published literature survival and neurodevelopmental outcome of fetuses with prenatally detected isolated severe bilateral ventriculomegaly.

METHODS

MEDLINE, EMBASE and the Cochrane Library were searched electronically. Only cases with a prenatal diagnosis of apparently isolated severe ventriculomegaly and postnatal neurodevelopmental assessment were selected and included. Severe ventriculomegaly was defined as enlargement of the ventricular atria, with a diameter of greater than 15 mm in the transventricular plane. All cases in which the investigators were unable to detect associated structural abnormality, chromosomal abnormality or fetal infection, and in which the ventriculomegaly was therefore regarded as apparently isolated, were included. Those for which the etiology was identified prenatally were excluded, whereas those with postnatal identification of the underlying cause were not excluded, since this information was not available prenatally. The quality of the included studies was assessed using the Newcastle-Ottawa Scale (NOS) for cohort studies. Pregnancy outcomes such as termination, stillbirth, neonatal survival and developmental outcome of the baby, were recorded. The degree of disability was classified as no, mild or severe disability. Statistical assessment was performed by meta-analysis of proportions to combine data, weighting the studies using the inverse variance method and a random-effects model. Proportions and CIs were reported.

RESULTS

Eleven studies including 137 fetuses were found. Twenty-seven pregnancies underwent termination and were excluded. The remaining 110 fetuses with apparently isolated severe ventriculomegaly for which continuation of pregnancy was intended, form the study population. Overall quality assessed using NOS for cohort studies was good. Survival was reported in 95/110 (pooled proportion 87.9% (95% CI, 75.6-96.2%)) cases. In 15/110 (pooled proportion 12.1% (95% CI, 3.8-24.4%)), either stillbirth or neonatal demise was reported. No disability was reported in 41/95 survivors (pooled proportion 42.2% (95% CI, 27.5-57.6%)). However, 17/95 showed mild/moderate disability (pooled proportion 18.6% (95% CI, 7.2-33.8%)) and 37/95 were reported to have severe disability (pooled proportion 39.6% (95% CI, 30.0-50.0%)).

CONCLUSIONS

Four-fifths of fetuses with severe ventriculomegaly survive and, of these, just over two-fifths show normal neurodevelopment. The overall survivors without disability account for more than one third of the total. Given that many cases undergo termination of pregnancy and require longer follow-up in order to detect subtle abnormalities, mortality and prevalence of developmental delay may be even higher than that reported in this paper. Copyright © 2018 ISUOG. Published by John Wiley & Sons Ltd.

摘要

目的

从已发表的文献中量化胎儿产前检测到的孤立性严重双侧脑室扩大的生存和神经发育结局。

方法

电子检索 MEDLINE、EMBASE 和 Cochrane 图书馆。仅选择和纳入具有产前诊断明显孤立性严重脑室扩大和产后神经发育评估的病例。严重脑室扩大定义为脑室心房扩张,在经脑室平面直径大于 15mm。所有研究者无法检测到相关结构异常、染色体异常或胎儿感染的病例,并且脑室扩大被认为是明显孤立性的,均被纳入。那些病因可在产前识别的病例被排除,而那些在产后确定潜在病因的病例则未被排除,因为这些信息在产前无法获得。使用纽卡斯尔-渥太华量表(NOS)评估纳入研究的质量。记录妊娠结局,如终止妊娠、死产、新生儿存活率和婴儿的发育结局。残疾程度分为无、轻度或重度残疾。通过合并数据的比例荟萃分析进行统计评估,使用逆方差法和随机效应模型对研究进行加权。报告了比例和 CIs。

结果

发现 11 项研究包括 137 例胎儿。27 例妊娠终止,被排除在外。其余 110 例具有明显孤立性严重脑室扩大且继续妊娠的胎儿构成了研究人群。使用 NOS 评估的队列研究整体质量良好。报告了 95/110(合并比例 87.9%(95%CI,75.6-96.2%))例的存活率。在 110/110 例(合并比例 12.1%(95%CI,3.8-24.4%))中,报告了死产或新生儿死亡。95 例存活者中无残疾(合并比例 42.2%(95%CI,27.5-57.6%))。然而,17/95 例表现为轻度/中度残疾(合并比例 18.6%(95%CI,7.2-33.8%)),37/95 例报告严重残疾(合并比例 39.6%(95%CI,30.0-50.0%))。

结论

四分之五的严重脑室扩大胎儿存活,其中超过五分之二的神经发育正常。无残疾的总体存活者占总数的三分之一以上。鉴于许多病例终止妊娠并需要更长时间的随访以发现细微异常,死亡率和发育迟缓的患病率可能甚至高于本文报道的水平。版权所有©2018 ISUOG。由 John Wiley & Sons Ltd 出版。

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