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胎儿颅内出血的围产期及长期结局:系统评价与荟萃分析

Perinatal and long-term outcome of fetal intracranial hemorrhage: systematic review and meta-analysis.

作者信息

Sileo F G, Zöllner J, D'Antonio F, Islam S, Papageorghiou A T, Khalil A

机构信息

Department of Biomedical, Metabolic and Neural Sciences, International Doctorate School in Clinical and Experimental Medicine, University of Modena and Reggio Emilia, Modena, Italy.

Unit of Obstetrics and Gynecology, Azienda Unità Sanitaria Locale - IRCCS, Reggio Emilia, Italy.

出版信息

Ultrasound Obstet Gynecol. 2022 May;59(5):585-595. doi: 10.1002/uog.24766.

DOI:10.1002/uog.24766
PMID:34529308
Abstract

OBJECTIVE

Fetal intracranial hemorrhage (ICH) is associated with an increased risk of perinatal mortality and morbidity. Healthcare professionals often find it challenging to counsel parents due to its rarity and diverse presentation. The aim of this systematic review and meta-analysis was to investigate the perinatal outcome of fetuses with ICH.

METHODS

MEDLINE, EMBASE, ClinicalTrials.gov and The Cochrane Library databases were searched. Inclusion criteria were studies reporting the outcome of fetuses, newborns and infants diagnosed with ICH. The primary outcome was perinatal death (PND), defined as the sum of intrauterine (IUD) and neonatal death (NND). The secondary outcomes were stillbirth, NND, IUD, termination of pregnancy, need for surgery/shunting at birth, cerebral palsy (defined according to the European Cerebral Palsy Network and classified as diplegia, hemiplegia, quadriplegia, dyskinetic or mixed), neurodevelopmental delay and intact survival. All outcomes were explored in the included fetuses with ICH. A subgroup analysis according to the location of the hemorrhage (intra-axial and extra-axial) was also planned. Meta-analysis of proportions was used to combine data, and pooled proportions and their 95% CI were reported.

RESULTS

Sixteen studies (193 fetuses) were included in the meta-analysis. PND occurred in 14.6% (95% CI, 7.3-24.0%) of fetuses with ICH. Among liveborn cases, 27.6% (95% CI, 12.5-45.9%) required shunt placement or surgery after birth and 32.0% (95% CI, 22.2-42.6%) had cerebral palsy. Furthermore, 16.7% (95% CI, 8.4-27.2%) of cases had mild neurodevelopmental delay, while 31.1% (95% CI, 19.0-44.7%) experienced severe adverse neurodevelopmental outcome. Normal neurodevelopmental outcome was reported in 53.6% of fetuses. Subgroup analysis according to the location of ICH showed that PND occurred in 13.3% (95% CI, 5.7-23.4%) of fetuses with intra-axial bleeding and 26.7% (95% CI, 5.3-56.8%) of those with extra-axial bleeding. In fetuses with intra-axial hemorrhage, 25.2% (95% CI, 11.0-42.9%) required shunt placement or surgery after birth and 25.5% (95% CI, 15.3-37.2%) experienced cerebral palsy. In fetuses with intra-axial hemorrhage, mild and severe neurodevelopmental delay was observed in 14.9% (95% CI, 12.0-27.0%) and 32.8% (95% CI, 19.8-47.4%) of cases, respectively, while 53.2% (95% CI, 37.0-69.1%) experienced normal neurodevelopmental outcome. The incidence of mortality and postnatal neurodevelopmental outcome in fetuses with extra-axial hemorrhage could not be estimated reliably due to the small number of cases.

CONCLUSIONS

Fetuses with a prenatal diagnosis of ICH are at high risk of perinatal mortality and adverse neurodevelopmental outcome. Postnatal shunt placement or surgery was required in 28% of cases and cerebral palsy was diagnosed in approximately one-third of infants. Due to the rarity of ICH, multicenter prospective registries are warranted to collect high-quality data. © 2021 International Society of Ultrasound in Obstetrics and Gynecology.

摘要

目的

胎儿颅内出血(ICH)与围产期死亡率和发病率升高相关。由于其罕见性和多样的表现形式,医疗保健专业人员常常发现向父母提供咨询具有挑战性。本系统评价和荟萃分析的目的是调查胎儿ICH的围产期结局。

方法

检索了MEDLINE、EMBASE、ClinicalTrials.gov和Cochrane图书馆数据库。纳入标准为报告诊断为ICH的胎儿、新生儿和婴儿结局的研究。主要结局为围产期死亡(PND),定义为宫内死亡(IUD)和新生儿死亡(NND)之和。次要结局为死产、NND、IUD、终止妊娠、出生时手术/分流需求、脑性瘫痪(根据欧洲脑性瘫痪网络定义并分类为双瘫、偏瘫、四肢瘫、运动障碍或混合型)、神经发育延迟和存活完好。在纳入的ICH胎儿中探讨所有结局。还计划根据出血部位(轴内和轴外)进行亚组分析。采用比例荟萃分析合并数据,并报告合并比例及其95%置信区间。

结果

16项研究(193例胎儿)纳入荟萃分析。ICH胎儿中PND发生率为14.6%(95%置信区间,7.3 - 24.0%)。在存活出生的病例中,27.6%(95%置信区间,12.5 - 45.9%)出生后需要进行分流或手术,32.0%(95%置信区间,22.2 - 42.6%)患有脑性瘫痪。此外,16.7%(95%置信区间,8.4 - 27.2%)的病例有轻度神经发育延迟,而31.1%(95%置信区间,19.0 - 44.7%)有严重不良神经发育结局。53.6%的胎儿报告神经发育结局正常。根据ICH部位进行的亚组分析显示,轴内出血胎儿中PND发生率为13.3%(95%置信区间,5.7 - 23.4%),轴外出血胎儿中为26.7%(95%置信区间,5.3 - 56.8%)。在轴内出血胎儿中,25.2%(95%置信区间,11.0 - 42.9%)出生后需要进行分流或手术,25.5%(95%置信区间,15.3 - 37.2%)患有脑性瘫痪。在轴内出血胎儿中,分别有14.9%(95%置信区间,12.0 - 27.0%)和32.8%(95%置信区间,19.8 - 47.4%)的病例有轻度和重度神经发育延迟,而53.2%(95%置信区间,37.0 - 69.1%)神经发育结局正常。由于病例数少,无法可靠估计轴外出血胎儿的死亡率和出生后神经发育结局发生率。

结论

产前诊断为ICH的胎儿围产期死亡率和不良神经发育结局风险高。28%的病例出生后需要进行分流或手术,约三分之一的婴儿被诊断为脑性瘫痪。由于ICH罕见,有必要建立多中心前瞻性登记处来收集高质量数据。© 2021国际妇产科超声学会。

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