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Pediatric cerebral cavernous malformations and stereotactic radiosurgery: an analysis of 50 cases from a multicentric study
Journal of Neurosurgery: Pediatrics ( IF 1.9 ) Pub Date : 2024-01-05 , DOI: 10.3171/2023.11.peds23402
Georgios Mantziaris 1 , Chloe Dumot 1, 2 , Stylianos Pikis 1 , Selcuk Peker 3 , Yavuz Samanci 3 , Gokce D. Ardor 3 , Ahmed M. Nabeel 4, 5 , Wael A. Reda 4, 6 , Sameh R. Tawadros 4, 6 , Khaled Abdel Karim 4, 7 , Amr M. N. El-Shehaby 4, 6 , Reem M. Emad Eldin 4, 8 , Ahmed H. Elazzazi 9 , Darrah Sheehan 1 , Kimball Sheehan 1 , Nuria Martínez Moreno 10 , Roberto Martínez Álvarez 10 , Roman Liscak 11 , Jaromir May 11 , Manjul Tripathi 6 , Akshay Rajput 6 , Narendra Kumar 12 , Rupinder Kaur 6 , Juan Diego Alzate 13 , Douglas Kondziolka 13 , Sam Dayawansa 1 , Jason P. Sheehan 1
Affiliation  

OBJECTIVE

Cerebral cavernous malformations (CCMs) are the second most common vascular anomaly affecting the CNS in children. Although stereotactic radiosurgery (SRS) has been proposed as an alternative to microsurgery in the management of selected cases in adults, there is a paucity of studies focusing on pediatric patients. The aim of this study was to present the outcomes and associated risks of SRS in this subgroup of patients.

METHODS

This retrospective multicenter study included pediatric patients treated with single-session SRS for CCMs. The annual hemorrhage rate (AHR) was calculated before and after SRS in hemorrhagic lesions. The Engel classification was used to describe post-SRS epileptic control. Adverse radiation effects (AREs) and the occurrence of new neurological deficits were recorded.

RESULTS

The study included 50 patients (median age 15.1 [IQR 5.6] years) harboring 62 CCMs. Forty-two (84%) and 22 (44%) patients had a history of hemorrhage or epilepsy prior to SRS, respectively. The AHR from diagnosis to SRS excluding the first hemorrhage was 7.19 per 100 CCM-years, dropping to 3.15 per 100 CCM-years after treatment. The cumulative risk of first hemorrhage after SRS was 7.4% (95% CI 0%–14.3%) at 5 years and 23.6% (95% CI 0%–42.2%) at 10 years. Eight hemorrhagic events involving 6 CCMs in 6 patients were recorded in the post-SRS follow-up period; 4 patients presented with transient symptoms and 4 with permanent symptoms. Of the 22 patients with pre-SRS seizures, 11 were seizure free at the last follow-up (Engel class I), 6 experienced improvement (Engel class II or III), 5 had no improvement (Engel class IVA or IVB), and 1 experienced worsening (Engel class IVC). Radiographic AREs were documented in 14.5% (9/62) of CCMs, with 4 being symptomatic.

CONCLUSIONS

Single-session SRS reduces the CCM hemorrhage rate in the pediatric population and provides adequate seizure control.



中文翻译:

小儿脑海绵状血管瘤与立体定向放射外科治疗:50例多中心研究分析

客观的

脑海绵状血管瘤 (CCM) 是影响儿童中枢神经系统的第二常见血管异常。尽管立体定向放射外科(SRS)已被提议作为显微外科手术的替代方案来治疗某些成人病例,但针对儿科患者的研究却很少。本研究的目的是展示该亚组患者接受放射外科手术的结果和相关风险。

方法

这项回顾性多中心研究包括接受单次 SRS 治疗 CCM 的儿科患者。计算出血性病变在 SRS 前后的年出血率 (AHR)。 Engel 分类用于描述 SRS 后癫痫控制。记录不良辐射效应(ARE)和新神经功能缺损的发生。

结果

该研究包括 50 名患有 62 个 CCM 的患者(中位年龄 15.1 [IQR 5.6] 岁)。 42 名 (84%) 和 22 名 (44%) 患者在接受 SRS 之前分别有出血或癫痫病史。从诊断到 SRS(不包括首次出血)的 AHR 为每 100 CCM 年 7.19,治疗后降至每 100 CCM 年 3.15。 SRS 术后首次出血的累积风险在 5 年时为 7.4% (95% CI 0%–14.3%),在 10 年时为 23.6% (95% CI 0%–42.2%)。 SRS 后随访期间记录了 6 名患者发生 8 起出血事件,涉及 6 个 CCM; 4 名患者出现短暂症状,4 名患者出现永久性症状。在 22 例 SRS 前癫痫发作的患者中,11 例在最后一次随访时癫痫发作消失(Engel I 级),6 例出现改善(Engel II 或 III 级),5 例无改善(Engel IVA 或 IVB 级), 1 名经历恶化(恩格尔 IVC 级)。 14.5% (9/62) 的 CCM 中记录有放射线 ARE,其中 4 例有症状。

结论

单次 SRS 可以降低儿科人群的 CCM 出血率,并提供充分的癫痫发作控制。

更新日期:2024-01-05
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