Ben-Israel David, Mann Jennifer A, Yang Michael M H, Isaacs Albert M, Cadieux Magalie, Sader Nicholas, Muram Sandeep, Albakr Abdulrahman, Manoranjan Branavan, Yu Richard W, Beland Benjamin, Hamilton Mark G, Spackman Eldon, Ronksley Paul E, Riva-Cambrin Jay
1Department of Clinical Neurosciences, University of Calgary.
2O'Brien Institute for Public Health, University of Calgary.
J Neurosurg Pediatr. 2022 May 6;30(1):18-30. doi: 10.3171/2022.3.PEDS21512. Print 2022 Jul 1.
Endoscopic third ventriculostomy and choroid plexus cauterization (ETV+CPC) is a novel procedure for infant hydrocephalus that was developed in sub-Saharan Africa to mitigate the risks associated with permanent implanted shunt hardware. This study summarizes the hydrocephalus literature surrounding the ETV+CPC intraoperative abandonment rate, perioperative mortality rate, cerebrospinal fluid infection rate, and failure rate.
This systematic review and meta-analysis followed a prespecified protocol and abides by Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. A comprehensive search strategy using MEDLINE, EMBASE, PsychInfo, Cochrane Central Register of Controlled Trials, Cochrane Database of Systematic Reviews, Scopus, and Web of Science was conducted from database inception to October 2019. Studies included controlled trials, cohort studies, and case-control studies of patients with hydrocephalus younger than 18 years of age treated with ETV+CPC. Pooled estimates were calculated using DerSimonian and Laird random-effects modeling, and the significance of subgroup analyses was tested using meta-regression. The quality of the pooled outcomes was assessed using the Grading of Recommendations Assessment, Development and Evaluation (GRADE) approach.
After screening and reviewing 12,321 citations, the authors found 16 articles that met the inclusion criteria. The pooled estimate for the ETV+CPC failure rate was 0.44 (95% CI 0.37-0.51). Subgroup analysis by geographic income level showed statistical significance (p < 0.01), with lower-middle-income countries having a lower failure rate (0.32, 95% CI 0.28-0.36) than high-income countries (0.53, 95% CI 0.47-0.60). No difference in failure rate was found between hydrocephalus etiology (p = 0.09) or definition of failure (p = 0.24). The pooled estimate for perioperative mortality rate (n = 7 studies) was 0.001 (95% CI 0.00-0.004), the intraoperative abandonment rate (n = 5 studies) was 0.04 (95% CI 0.01-0.08), and the postoperative CSF infection rate (n = 5 studies) was 0.0004 (95% CI 0.00-0.003). All pooled outcomes were found to be low-quality evidence.
This systematic review and meta-analysis provides the most comprehensive pooled estimate for the ETV+CPC failure rate to date and demonstrates, for the first time, a statistically significant difference in failure rate by geographic income level. It also provides the first reported pooled estimates for the risk of ETV+CPC perioperative mortality, intraoperative abandonment, and CSF infection. The low quality of this evidence highlights the need for further research to improve the understanding of these critical clinical outcomes and their relevant explanatory variables and thus to appreciate which patients may benefit most from an ETV+CPC. Systematic review registration no.: CRD42020160149 (https://www.crd.york.ac.uk/prospero/).
内镜下第三脑室造瘘术和脉络丛烧灼术（ETV+CPC）是一种针对婴儿脑积水的新手术，该手术是在撒哈拉以南非洲地区开发的，以降低与永久性植入分流硬件相关的风险。本研究总结了围绕ETV+CPC术中放弃率、围手术期死亡率、脑脊液感染率和失败率的脑积水相关文献。
本系统评价和荟萃分析遵循预先指定的方案，并遵守系统评价和荟萃分析的首选报告项目（PRISMA）指南。从数据库建立到2019年10月，使用MEDLINE、EMBASE、PsychInfo、Cochrane对照试验中央注册库、Cochrane系统评价数据库、Scopus和科学网进行了全面的检索策略。纳入的研究包括对18岁以下接受ETV+CPC治疗的脑积水患者的对照试验、队列研究和病例对照研究。使用DerSimonian和Laird随机效应模型计算合并估计值，并使用meta回归检验亚组分析的显著性。使用推荐分级评估、制定和评价（GRADE）方法评估合并结果的质量。
在筛选和审查了12321篇文献后，作者发现16篇文章符合纳入标准。ETV+CPC失败率的合并估计值为0.44（95%CI 0.37-0.51）。按地理收入水平进行的亚组分析显示具有统计学显著性（p<0.01），中低收入国家的失败率（0.32，95%CI 0.28-0.36）低于高收入国家（0.53，95%CI 0.47-0.60）。在脑积水病因（p=0.09）或失败定义（p=0.24）之间未发现失败率有差异。围手术期死亡率（n=7项研究）的合并估计值为0.001（95%CI 0.00-0.004），术中放弃率（n=5项研究）为0.04（95%CI 0.01-0.08），术后脑脊液感染率（n=5项研究）为0.0004（95%CI 0.00-0.003）。所有合并结果均为低质量证据。
本系统评价和荟萃分析提供了迄今为止ETV+CPC失败率最全面的合并估计值，并首次证明按地理收入水平的失败率存在统计学显著性差异。它还首次报告了ETV+CPC围手术期死亡率、术中放弃和脑脊液感染风险的合并估计值。该证据的低质量凸显了进一步研究的必要性，以增进对这些关键临床结果及其相关解释变量的理解，从而了解哪些患者可能从ETV+CPC中获益最大。系统评价注册号：CRD42020160149（https://www.crd.york.ac.uk/prospero/）。
