Departments of1Neurological Surgery and.
2Radiology; and.
J Neurosurg. 2021 Jul 30;136(2):492-502. doi: 10.3171/2021.1.JNS203476. Print 2022 Feb 1.
Cerebral arteriovenous malformations (AVMs) are rare cerebral vascular lesions that are associated with high morbidity and mortality from hemorrhage; however, stereotactic radiosurgery (SRS) is a well-validated treatment modality. Few reports have delineated a subgroup of patients who develop delayed chronic encapsulated expanding hematomas (CEEHs) despite angiographic evidence of AVM obliteration following radiosurgery. In this report, the authors performed a retrospective review of more than 1000 radiosurgically treated intracranial AVM cases to delineate the incidence and management of this rare entity.
Between 1988 and 2019, 1010 patients with intracranial AVM underwent Gamma Knife SRS at the University of Pittsburgh Medical Center. In addition to a review of a prospective institutional database, the authors performed a retrospective chart review of the departmental AVM database to specifically identify patients with CEEH. Pertinent clinical and radiological characteristics as well as patient outcomes were recorded and analyzed.
Nine hundred fifty patients with intracranial AVM (94%) had sufficient clinical follow-up for analysis. Of these, 6 patients with CEEH underwent delayed resection (incidence rate of 0.0045 event per person-year). These patients included 4 males and 2 females with a mean age of 45.3 ± 13.8 years at the time of initial SRS. Four patients had smaller AVM volumes (4.9-10 cm3), and 3 of them were treated with a single SRS procedure. Two patients had larger-volume AVMs (55 and 56 cm3), and both underwent multimodal management that included staged SRS and embolization. Time to initial recognition of the CEEH after initial SRS ranged between 66 and 243 months. The time between CEEH recognition and resection ranged from 2 to 9 months. Resection was required because of progressive neurological symptoms that correlated with imaging evidence of gradual hematoma expansion. All 6 patients had angiographically confirmed obliteration of their AVM. Pathology revealed a mixed chronicity hematoma with areas of fibrosed blood vessels and rare areas of neovascularization with immature blood vessels but no evidence of a persistent AVM. All 6 patients reported persistent clinical improvement after hematoma resection.
CEEH after SRS for AVM is a rare complication with an incidence rate of 0.0045 event per person-year over the authors' 30-year experience. When clinical symptoms progress and imaging reveals progressive enlargement over time, complete resection of a CEEH results in significant clinical recovery. Knowledge of this rare entity facilitates timely detection and eventual surgical intervention to achieve optimal outcomes.
脑动静脉畸形(AVM)是一种罕见的脑血管病变,其出血导致的发病率和死亡率很高;然而,立体定向放射外科(SRS)是一种经过充分验证的治疗方法。尽管血管造影显示 SRS 后 AVM 闭塞,但很少有报道描述了一组患者会出现延迟性慢性包裹性扩张性血肿(CEEH)。在本报告中,作者对 1000 多例接受放射外科治疗的颅内 AVM 病例进行了回顾性分析,以阐明这种罕见实体的发生率和治疗方法。
1988 年至 2019 年期间,匹兹堡大学医学中心的 1010 例颅内 AVM 患者接受了伽玛刀 SRS。除了对一个前瞻性机构数据库进行回顾外,作者还对部门 AVM 数据库进行了回顾性图表审查,以专门确定患有 CEEH 的患者。记录并分析了相关的临床和影像学特征以及患者结局。
950 例颅内 AVM 患者(94%)有足够的临床随访进行分析。其中,6 例患者发生了迟发性 CEEH,需要进行延迟性切除(0.0045 例/人年的发生率)。这些患者包括 4 名男性和 2 名女性,在初次 SRS 时的平均年龄为 45.3±13.8 岁。4 例患者的 AVM 体积较小(4.9-10cm3),其中 3 例接受单次 SRS 治疗。2 例患者的 AVM 体积较大(55 和 56cm3),均接受了包括分期 SRS 和栓塞的多模态治疗。初次 SRS 后初次发现 CEEH 的时间为 66-243 个月。从 CEEH 发现到切除的时间为 2-9 个月。进行切除是因为神经症状逐渐加重,影像学显示血肿逐渐扩大。所有 6 例患者的 AVM 均经血管造影证实完全闭塞。病理显示为混合慢性血肿,伴有纤维化血管和罕见的不成熟血管的新生血管区域,但没有持续存在的 AVM 的证据。所有 6 例患者在血肿切除后均报告持续的临床改善。
SRS 治疗 AVM 后发生 CEEH 是一种罕见的并发症,作者在 30 年的经验中,其发生率为 0.0045 例/人年。当临床症状进展且影像学显示随时间逐渐增大时,完全切除 CEEH 可显著改善临床结局。了解这种罕见实体有助于及时发现并最终进行手术干预,以实现最佳结局。