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先天性膈疝患儿的神经发育损害:幸存者并不少见的并发症。

Neurodevelopmental impairment in children with congenital diaphragmatic hernia: Not an uncommon complication for survivors.

机构信息

Division of General and Thoracic Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Canada.

Division of General and Thoracic Surgery, The Hospital for Sick Children, University of Toronto, Toronto, Canada; Department of Pediatric Surgery, "Spirito Santo" Hospital, Pescara, and "G. d'Annunzio" University, Chieti-Pescara, Italy.

出版信息

J Pediatr Surg. 2020 Apr;55(4):625-634. doi: 10.1016/j.jpedsurg.2019.05.021. Epub 2019 Jun 7.

Abstract

PURPOSE

To evaluate neurodevelopmental impairment (NDI) in children born with congenital diaphragmatic hernia (CDH).

METHODS

Using a defined search strategy, a systematic review was conducted to define the incidence and types of NDI, to report abnormal neuroimaging findings and to evaluate possible NDI predictors. A meta-analysis was performed on comparative studies reporting risk factors for NDI, using RevMan 5.3.

RESULTS

Of 3541 CDH children (33 studies), 829 (23%) had NDI, with a higher incidence in CDH survivors who received ECMO treatment (49%) vs. those who had no ECMO (22%; p<0.00001). NDI included neuromuscular hypotonia (42%), hearing (13%) and visual (8%) impairment, neurobehavioral issues (20%), and learning difficulties (31%). Of 288 survivors that had postnatal neuroimaging, 49% had abnormal findings. The main risk factors for NDI were severe pulmonary hypoplasia, large defect size, ECMO use.

CONCLUSIONS

NDI is a relevant problem for CDH survivors, affecting 1 in 4. The spectrum of NDI covers all developmental domains and ranges from motor and sensory (hearing, visual) deficits to cognitive, language, and behavioral impairment. Further studies should be designed to better understand the pathophysiology of NDI in CDH children and to longitudinally monitor infants born with CDH to correct risk factors that can be modifiable.

LEVEL OF EVIDENCE

Level III.

摘要

目的

评估先天性膈疝(CDH)患儿的神经发育障碍(NDI)。

方法

使用明确的搜索策略,进行系统评价以确定 NDI 的发生率和类型,报告异常神经影像学发现,并评估可能的 NDI 预测因素。使用 RevMan 5.3 对报告 NDI 危险因素的比较研究进行荟萃分析。

结果

在 3541 例 CDH 患儿(33 项研究)中,829 例(23%)存在 NDI,接受 ECMO 治疗的 CDH 幸存者发生率更高(49%),而未接受 ECMO 治疗的患儿发生率较低(22%;p<0.00001)。NDI 包括神经肌肉张力减退(42%)、听力(13%)和视力(8%)损害、神经行为问题(20%)和学习困难(31%)。在 288 例接受产后神经影像学检查的幸存者中,49%存在异常发现。NDI 的主要危险因素是严重肺发育不良、大的缺损大小、ECMO 使用。

结论

NDI 是 CDH 幸存者的一个相关问题,影响 1/4 的患儿。NDI 的谱系涵盖所有发育领域,从运动和感觉(听力、视力)缺陷到认知、语言和行为障碍。应设计进一步的研究以更好地了解 CDH 患儿 NDI 的病理生理学,并对出生时患有 CDH 的婴儿进行纵向监测,以纠正可改变的危险因素。

证据水平

III 级。

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