小儿脑海绵状血管畸形与立体定向放射外科治疗：一项多中心研究中50例病例分析
Pediatric cerebral cavernous malformations and stereotactic radiosurgery: an analysis of 50 cases from a multicentric study.
作者信息
Mantziaris Georgios, Dumot Chloe, Pikis Stylianos, Peker Selcuk, Samanci Yavuz, Ardor Gokce D, Nabeel Ahmed M, Reda Wael A, Tawadros Sameh R, Abdel Karim Khaled, El-Shehaby Amr M N, Emad Eldin Reem M, Elazzazi Ahmed H, Sheehan Darrah, Sheehan Kimball, Martínez Moreno Nuria, Martínez Álvarez Roberto, Liscak Roman, May Jaromir, Tripathi Manjul, Rajput Akshay, Kumar Narendra, Kaur Rupinder, Alzate Juan Diego, Kondziolka Douglas, Dayawansa Sam, Sheehan Jason P
机构信息
1Department of Neurological Surgery, University of Virginia, Charlottesville, Virginia.
2Department of Neurological Surgery, Hospices Civils de Lyon, France.
出版信息
J Neurosurg Pediatr. 2024 Jan 5;33(4):315-322. doi: 10.3171/2023.11.PEDS23402. Print 2024 Apr 1.
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.
目的
脑海绵状血管畸形（CCM）是影响儿童中枢神经系统（CNS）的第二常见血管异常。尽管立体定向放射外科手术（SRS）已被提议作为成人特定病例显微手术的替代方案，但针对儿科患者的研究较少。本研究的目的是介绍该亚组患者接受SRS的结果及相关风险。
方法
这项回顾性多中心研究纳入了接受单疗程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）。采用恩格尔分类法描述SRS后癫痫控制情况。记录不良放射效应（AREs）和新神经功能缺损的发生情况。
结果
该研究纳入了50例患者（中位年龄15.1[四分位间距5.6]岁），共62个CCM。分别有42例（84%）和22例（44%）患者在SRS前有出血或癫痫病史。从诊断到SRS（不包括首次出血）的AHR为每100个CCM年7.19次，治疗后降至每100个CCM年3.15次。SRS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%）。在SRS后的随访期内记录到8次出血事件，涉及6例患者的6个CCM；4例患者出现短暂症状，4例出现永久性症状。在22例SRS前有癫痫发作的患者中，11例在最后一次随访时无癫痫发作（恩格尔I级），6例病情改善（恩格尔II级或III级），5例无改善（恩格尔IVA或IVB级），1例病情恶化（恩格尔IVC级）。62个CCM中有14.5%（9/62）记录到影像学AREs，其中4个有症状。
结论
单疗程SRS可降低儿科患者CCM的出血率，并能充分控制癫痫发作。
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