Department of Diagnostic Imaging, The Hospital for Sick Children, University of Toronto, 555 University Ave, Toronto, ON M5G 1X8, Canada.
NeuroSpine Research Group, Liverpool Hospital, Sydney, Australia.
AJR Am J Roentgenol. 2020 Aug;215(2):472-487. doi: 10.2214/AJR.19.22443. Epub 2020 Jun 6.
The objective of this study was to assess the available evidence in the literature regarding treatment outcomes for pediatric patients with brain arteriovenous malformation (bAVM) with the aim of providing practice guidelines for treatment decisions and highlighting research areas that need attention. Keyword searches for studies published from January 1, 1981, to April 16, 2018, were performed in MEDLINE, Embase, and Web of Science. Predefined inclusion criteria were used to identify studies. Poisson regression analysis for associations between patient and bAVM characteristics and treatment outcomes. We identified 34 articles comprising 2158 children with bAVM who underwent treatment or observation. The mean age of the study cohort was 12.0 ± 1.6 (SD) years, and 48.1% of the patients were female; 64.3% of bAVMs were hemorrhagic at presentation. The mean follow-up was 50.6 ± 32.3 months. Overall, the meta-analysis of pooled data showed an obliteration rate of 69.8% (95% CI, 62.9-75.9%), recurrence rate of 2.2% (95% CI, 1.1-4.3%), and mortality rate of 2.4%. The pooled complication rate was 22.5% (95% CI, 15.7-31.1%) after surgery, 26.4% (95% CI, 15.2-41.9%) after embolization, and 27.1% (95% CI, 18.1-38.4%) after radiosurgery. Mortality was not associated with age, sex, or hemorrhage; however, recurrence after treatment was inversely associated with age. Complication and mortality rates were reduced for multimodal treatments. For patients with bAVM treated with observation only, complication and mortality rates were 35.9% and 23.5%, respectively. Multimodality treatments for pediatric bAVM had lower mortality and complication rates than individual treatments. However, there is a lack of evidence for long-term outcomes. The mortality rate was highest in conservatively managed patients (i.e., observation only). Further research directly comparing different treatment modalities for recurrence and complications is warranted. Gathering data prospectively through multiinstitutional registries will be key to provide strong evidence.
本研究旨在评估文献中关于儿童脑动静脉畸形(bAVM)治疗结果的现有证据,旨在为治疗决策提供实践指南,并强调需要关注的研究领域。1981 年 1 月 1 日至 2018 年 4 月 16 日,在 MEDLINE、Embase 和 Web of Science 中进行了针对已发表研究的关键字搜索。使用预定义的纳入标准来识别研究。采用泊松回归分析患者和 bAVM 特征与治疗结果之间的关联。我们确定了 34 篇文章,共纳入 2158 名接受治疗或观察的 bAVM 患儿。研究队列的平均年龄为 12.0±1.6(标准差)岁,48.1%的患者为女性;64.3%的 bAVM 在就诊时为出血性。平均随访时间为 50.6±32.3 个月。总体而言,汇总数据的荟萃分析显示闭塞率为 69.8%(95%CI,62.9-75.9%),复发率为 2.2%(95%CI,1.1-4.3%),死亡率为 2.4%。手术、栓塞和放射外科治疗后的并发症发生率分别为 22.5%(95%CI,15.7-31.1%)、26.4%(95%CI,15.2-41.9%)和 27.1%(95%CI,18.1-38.4%)。死亡率与年龄、性别或出血无关;然而,治疗后复发与年龄呈负相关。多模式治疗可降低并发症和死亡率。对于仅接受观察治疗的 bAVM 患者,并发症和死亡率分别为 35.9%和 23.5%。与单一治疗相比,儿童 bAVM 的多模式治疗具有较低的死亡率和并发症发生率。然而,长期结果的证据不足。保守治疗(即仅观察)患者的死亡率最高。有必要进行直接比较不同治疗方式的复发和并发症的研究。通过多机构登记处前瞻性收集数据将是提供有力证据的关键。