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小儿动静脉畸形的立体定向放射外科治疗:一项PRISMA系统评价和荟萃分析。

Stereotactic radiosurgery treatment of pediatric arteriovenous malformations: a PRISMA systematic review and meta-analysis.

作者信息

Thrash Garrett W, Evans Riley Ethan, Sun Yifei, Roberts Anne C, Derryberry Cameron, Hale Andrew T, Das Somnath, Boudreau Hunter, George Jordan A, Atchley Travis J, Blount Jeffrey P, Rocque Brandon G, Johnston James M, Jones Jesse G

机构信息

Heersink School of Medicine, University of Alabama at Birmingham, FOT Suite, 1720 2nd Ave S, Birmingham, AL, 35294, USA.

Department of Neurosurgery, University of Alabama at Birmingham, Birmingham, AL, USA.

出版信息

Childs Nerv Syst. 2025 May 23;41(1):188. doi: 10.1007/s00381-025-06835-z.

Abstract

BACKGROUND

Stereotactic radiosurgery (SRS) is considered a safe definitive treatment for pediatric arteriovenous malformations (AVMs) upon indicated presentations. There are no published guidelines by the International Stereotactic Radiosurgery Society (ISRS) detailed with indications or characteristics that warrant SRS, other than the guideline that SRS is a safe and efficacious treatment for pediatric AVMs. SRS is performed using either Gamma Knife (GK) or Linear Accelerator (LINAC). This systematic review aims to uncover treatment, differences in GK and LINAC outcomes, and AVM characteristics that lead to high obliteration rates and suggest future studies to determine treatment decisions, raise obliteration rates, and lower complication rates in SRS treatment of pediatric AVMs.

METHODS

We performed a systematic review according to PRISMA guidelines across PubMed, Embase, and SCOPUS utilizing search terms related to pediatric patients, AVMS, and SRS. We collected data from the 32 full-text studies and 4 abstracts that met inclusion criteria. Subsequent pooled analysis was performed on GK vs LINAC obliteration rates, followed by sub-cohort analysis of all SRS patients with hemorrhagic presentation, Spetzler-Martin (SM) Grade, and prior procedure and their effect on obliteration rates.

RESULTS

The 36 studies reported 3425 patients, with a slight male preponderance (1662 patients, 48.5%). The obliteration analysis included 2834 patients that met follow-up criteria and contained obliteration data. The weighted mean age was 12.63 years. Pooled cohort analysis found no significant difference in obliteration proportions when comparing GK to LINAC (P = 0.7449), with an overall obliteration rate of 63% in patients with at least 1 year follow-up. The sub-cohort analysis of all patients treated with SRS revealed that presentation with AVM hemorrhage was associated with increased obliteration (CE: RR = 1.22 [95%CI = 1.09-1.35; RE: RR = 1.22, 95%CI = 10.6-1.40; prediction interval = 1.07-1.38) with low heterogeneity (I = 17.1%, τ < 0.0001, p = 0.2902). Smaller SM grade was not statistically associated with increased obliteration (CE: RR = 1.25 [95%CI = 0.87-1.81]; RE: RR = 1.84 [95%CI = 0.97-3.50]; prediction interval = 0.38-8.86) and moderate levels of heterogeneity were detected (I = 45.2%, τ = 0.2668, p = 0.1042). Procedure prior to SRS also had higher obliteration rates than no prior procedure (CE: RR = 0.77 [95%CI = 0.61-0.86]; RE: RR = 0.71 [95%CI = 0.54-0.92]; prediction interval = 0.36-1.39) with low to moderate heterogeneity (I = 27.6%, τ = 0.0.0264, p = 0.2466).

CONCLUSION

SRS is a safe and effective treatment for pediatric AVMs. This study suggests that there are no differences in obliteration between GK and LINAC, with increased obliteration in patients with hemorrhage at presentation and procedures prior to SRS treatment. Further multicenter, prospective studies are necessary to dictate future treatment decisions.

摘要

背景

立体定向放射外科手术(SRS)被认为是治疗小儿动静脉畸形(AVM)的一种安全的确定性治疗方法。除了SRS是治疗小儿AVM的一种安全有效的治疗方法这一指南外,国际立体定向放射外科协会(ISRS)没有发布详细说明SRS适应证或特征的指南。SRS可使用伽玛刀(GK)或直线加速器(LINAC)进行。本系统评价旨在揭示治疗方法、GK和LINAC治疗效果的差异,以及导致高闭塞率的AVM特征,并建议未来开展研究以确定治疗决策,提高SRS治疗小儿AVM的闭塞率并降低并发症发生率。

方法

我们根据PRISMA指南,在PubMed、Embase和SCOPUS上进行了系统评价,使用了与小儿患者、AVM和SRS相关的检索词。我们从32篇全文研究和4篇摘要中收集了符合纳入标准的数据。随后对GK和LINAC的闭塞率进行了汇总分析,接着对所有有出血表现、Spetzler-Martin(SM)分级、既往治疗史及其对闭塞率影响的SRS患者进行了亚组分析。

结果

36项研究报告了3425例患者,男性略占优势(1662例患者,48.5%)。闭塞分析纳入了2834例符合随访标准且有闭塞数据的患者。加权平均年龄为12.63岁。汇总队列分析发现,比较GK和LINAC时,闭塞比例无显著差异(P = 0.7449),至少随访1年的患者总体闭塞率为63%。对所有接受SRS治疗的患者进行的亚组分析显示,AVM出血表现与闭塞率增加相关(CE:RR = 1.22 [95%CI = 1.09 - 1.35;RE:RR = 1.22,95%CI = 10.6 - 1.40;预测区间 = 1.07 - 1.38]),异质性较低(I = 17.1%,τ < 0.0001,p = 0.2902)。较小的SM分级与闭塞率增加无统计学关联(CE:RR = 1.25 [95%CI = 0.87 - 1.81];RE:RR = 1.84 [95%CI = 0.97 - 3.50];预测区间 = 0.38 - 8.86),检测到中等程度的异质性(I = 45.2%,τ = 0.2668,p = 0.1042)。SRS治疗前有过治疗的患者闭塞率也高于未进行过治疗的患者(CE:RR = 0.77 [95%CI = 0.61 - 0.86];RE:RR = 0.71 [95%CI = 0.54 - 0.92];预测区间 = 0.36 - 1.39),异质性低至中等(I = 27.6%,τ = 0.0264,p = 0.2466)。

结论

SRS是治疗小儿AVM的一种安全有效的方法。本研究表明,GK和LINAC在闭塞率方面无差异,出血表现的患者以及SRS治疗前有过治疗的患者闭塞率增加。需要进一步开展多中心前瞻性研究来指导未来的治疗决策。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6cf/12102109/2bba71c2cb24/381_2025_6835_Fig1_HTML.jpg

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