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小儿持续性脑动静脉畸形的重复立体定向放射外科治疗

Repeat stereotactic radiosurgery for persistent cerebral arteriovenous malformations in pediatric patients.

作者信息

Garcia Gracie, Mantziaris Georgios, Pikis Stylianos, Dumot Chloe, Lunsford L Dade, Niranjan Ajay, Wei Zhishuo, Srinivasan Priyanka, Tang Lilly W, Liscak Roman, May Jaromir, Lee Cheng-Chia, Yang Huai-Che, Peker Selcuk, Samanci Yavuz, Nabeel Ahmed M, Reda Wael A, Tawadros Sameh R, Abdel Karim Khaled, El-Shehaby Amr M N, Emad Eldin Reem, Elazzazi Ahmed Hesham, Martínez Moreno Nuria, Martínez Álvarez Roberto, Padmanaban Varun, Jareczek Francis J, McInerney James, Cockroft Kevin M, Alzate Juan Diego, Kondziolka Douglas, Tripathi Manjul, Sheehan Jason P

机构信息

1Department of Neurological Surgery, University of Virginia, Charlottesville, Virginia.

2Department of Neurological Surgery, University of Pittsburgh Medical Center, Pennsylvania.

出版信息

J Neurosurg Pediatr. 2024 Jan 26;33(4):307-314. doi: 10.3171/2023.12.PEDS23465. Print 2024 Apr 1.

Abstract

OBJECTIVE

The purpose of this study was to describe the long-term outcomes and associated risks related to repeat stereotactic radiosurgery (SRS) for persistent arteriovenous malformations (AVMs) in pediatric patients.

METHODS

Under the auspices of the International Radiosurgery Research Foundation, this retrospective multicenter study analyzed pediatric patients who underwent repeat, single-session SRS between 1987 and 2022. The primary outcome variable was a favorable outcome, defined as nidus obliteration without hemorrhage or neurological deterioration. Secondary outcomes included rates and probabilities of hemorrhage, radiation-induced changes (RICs), and cyst or tumor formation.

RESULTS

The cohort included 83 pediatric patients. The median patient age was 11 years at initial SRS and 15 years at repeat SRS. Fifty-seven children (68.7%) were managed exclusively using SRS, and 42 (50.6%) experienced hemorrhage prior to SRS. Median AVM diameter and volume were substantially different between the first (25 mm and 4.5 cm3, respectively) and second (16.5 mm and 1.6 cm3, respectively) SRS, while prescription dose and isodose line remained similar. At the 5-year follow-up evaluation from the second SRS, nidus obliteration was achieved in 42 patients (50.6%), with favorable outcome in 37 (44.6%). The median time to nidus obliteration and hemorrhage was 35.5 and 38.5 months, respectively. The yearly cumulative probability of favorable outcome increased from 2.5% (95% CI 0.5%-7.8%) at 1 year to 44% (95% CI 32%-55%) at 5 years. The probability of achieving obliteration followed a similar pattern and reached 51% (95% CI 38%-62%) at 5 years. The 5-year risk of hemorrhage during the latency period after the second SRS reached 8% (95% CI 3.2%-16%). Radiographically, 25 children (30.1%) had RICs, but only 5 (6%) were symptomatic. Delayed cyst formation occurred in 7.2% of patients, with a median onset of 47 months. No radiation-induced neoplasia was observed.

CONCLUSIONS

The study results showed nidus obliteration in most pediatric patients who underwent repeat SRS for persistent AVMs. The risks of symptomatic RICs and latency period hemorrhage were quite low. These findings suggest that repeat radiosurgery should be considered when treating pediatric patients with residual AVM after prior SRS. Further study is needed to define the role of repeat SRS more fully in this population.

摘要

目的

本研究旨在描述小儿持续性动静脉畸形(AVM)重复立体定向放射外科治疗(SRS)的长期疗效及相关风险。

方法

在国际放射外科研究基金会的支持下,这项回顾性多中心研究分析了1987年至2022年间接受重复单次SRS治疗的小儿患者。主要结局变量为良好结局,定义为病灶闭塞且无出血或神经功能恶化。次要结局包括出血率和概率、放射诱导变化(RICs)以及囊肿或肿瘤形成情况。

结果

该队列包括83例小儿患者。初次SRS时患者的中位年龄为11岁,重复SRS时为15岁。57名儿童(68.7%)仅接受SRS治疗,42名(50.6%)在SRS前发生过出血。初次SRS与第二次SRS时AVM的中位直径和体积有显著差异,分别为25 mm和4.5 cm³,以及16.5 mm和1.6 cm³,而处方剂量和等剂量线保持相似。在第二次SRS后的5年随访评估中,42例患者(50.6%)实现了病灶闭塞,37例(44.6%)获得了良好结局。病灶闭塞和出血的中位时间分别为35.5个月和38.5个月。良好结局的年累积概率从1年时的2.5%(95%CI 0.5%-7.8%)增至5年时的44%(95%CI 32%-55%)。实现闭塞的概率遵循类似模式,5年时达到51%(95%CI 38%-62%)。第二次SRS后潜伏期内5年的出血风险达到8%(95%CI 3.2%-16%)。影像学检查显示,25名儿童(30.1%)有RICs,但只有5名(6%)有症状。7.2%的患者发生延迟性囊肿形成,中位发病时间为47个月。未观察到放射诱导的肿瘤。

结论

研究结果表明,大多数接受重复SRS治疗持续性AVM的小儿患者实现了病灶闭塞。有症状的RICs和潜伏期出血的风险相当低。这些发现表明,对于先前接受SRS后仍有残留AVM的小儿患者,治疗时应考虑重复放射外科治疗。需要进一步研究以更全面地确定重复SRS在该人群中的作用。

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