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小儿脑深部动静脉畸形的自然史和治疗。

Natural history and treatment of deep-seated brain arteriovenous malformations in pediatric patients.

出版信息

J Neurosurg Pediatr. 2022 Sep 9;30(6):578-585. doi: 10.3171/2022.8.PEDS22213. Print 2022 Dec 1.

Abstract

OBJECTIVE

Pediatric deep brain arteriovenous malformations (bAVMs) represent a unique management challenge given their higher cumulative risk of hemorrhage as well as a higher risk of treatment. Better understanding of hemorrhage risk in this patient population will lead to a better decision-making process for patient management.

METHODS

The authors retrospectively reviewed their institutional bAVM database from 1990 to 2019 and included patients younger than 21 years who had deep-seated bAVMs. They present the annual hemorrhage risk, during the natural history and after treatment, and functional outcomes.

RESULTS

Thirty-one pediatric patients were included in this study (13 males and 18 females) with a mean age of 11.8 (SD 4.4) years. The most frequent presenting symptoms were headache (54.8%), weakness (38.7%), and seizure (22.6%). The mean follow-up duration was 13.14 (SD 12.5) years, during which 7 (22.6%) AVMs were obliterated, 10 (32.3%) individuals experienced hemorrhage, and the modified Rankin Scale score worsened in 8 (25.8%) patients. The annual natural history risk of hemorrhage was 3.24% per patient, and the overall annual hemorrhage risk after treatment was 1.98% per patient. In particular, the risk was reduced to 0.64% per patient in the stereotactic radiosurgery (SRS) group. Non-White race showed a trend of higher rupture at presentation (OR 5 [95% CI 0.84-41.68], p = 0.09). Female sex was associated with higher odds (OR 13.076 [95% CI 1.424-333.591], p = 0.048) and SRS was associated with lower odds (OR 0.122 [95% CI 0.011-0.862], p = 0.049) of follow-up hemorrhage.

CONCLUSIONS

Given the substantial cumulative risk of lifelong hemorrhagic stroke in pediatric patients, timely definitive treatment is warranted. SRS may be beneficial when the risk-benefit profile is deemed acceptable.

摘要

目的

由于儿科深部脑动静脉畸形(bAVM)的出血累积风险较高,且治疗风险也较高,因此在管理方面具有独特的挑战性。更好地了解该患者人群的出血风险将有助于制定更好的患者管理决策。

方法

作者回顾性分析了其机构从 1990 年至 2019 年的 bAVM 数据库,纳入了年龄小于 21 岁且存在深部 bAVM 的患者。作者呈现了自然史期间和治疗后的年度出血风险,以及功能结局。

结果

本研究纳入了 31 名儿科患者(男性 13 名,女性 18 名),平均年龄为 11.8(SD 4.4)岁。最常见的首发症状是头痛(54.8%)、无力(38.7%)和癫痫发作(22.6%)。平均随访时间为 13.14(SD 12.5)年,在此期间,7 例(22.6%)AVM 闭塞,10 例(32.3%)患者发生出血,8 例(25.8%)患者的改良 Rankin 量表评分恶化。每年自然史出血风险为每位患者 3.24%,治疗后每年出血风险为每位患者 1.98%。特别是,在立体定向放射外科(SRS)组中,风险降低至每位患者 0.64%。非白种人在就诊时更易发生破裂(OR 5[95%CI 0.84-41.68],p=0.09)。女性的出血几率更高(OR 13.076[95%CI 1.424-333.591],p=0.048),SRS 的出血几率更低(OR 0.122[95%CI 0.011-0.862],p=0.049)。

结论

鉴于儿科患者终生出血性中风的累积风险较大,因此需要及时进行确定性治疗。当风险效益比被认为可以接受时,SRS 可能是有益的。

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