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桥脑旁动静脉畸形伴三叉神经痛患者的治疗结果。

Management outcomes of peripontine arteriovenous malformation patients presenting with trigeminal neuralgia.

机构信息

1Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland; and.

2Department of Neurosurgery, Stanford University School of Medicine, Stanford, California.

出版信息

J Neurosurg. 2023 Jul 14;140(2):515-521. doi: 10.3171/2023.5.JNS23771. Print 2024 Feb 1.

DOI:10.3171/2023.5.JNS23771
PMID:37486910
Abstract

OBJECTIVE

Trigeminal neuralgia as the presenting symptom of brain arteriovenous malformation (bAVM) has been rarely reported. Treatment of reported cases has been skewed toward surgery for these scarce, deeply located bAVMs. Here, the authors report their management and outcomes of bAVM patients presenting with ipsilateral trigeminal neuralgia (TN) at their institution.

METHODS

This is a retrospective cohort study. The authors' institutional bAVM database was queried for non-hereditary hemorrhagic telangiectasia bAVMs in pontine, cistern, brainstem, trigeminal nerve, or tentorial locations. Patients with complete data were included in a search for trigeminal neuralgia or "facial pain" as the presenting symptom with TN being on the same side as the bAVM. Demographics, TN and bAVM characteristics, management strategies, and outcomes of bAVM and TN management were analyzed.

RESULTS

Fifty-seven peripontine bAVMs were identified; 8 (14.0%) of these bAVMs were discovered because of ipsilateral TN, including 4 patients (50%) with facial pain in the V2 distribution. Five patients (62.5%) were treated with carbamazepine as the initial medical therapy, 2 (25%) underwent multiple rhizotomies, and 1 (12.5%) underwent microvascular decompression. None of the patients with TN-associated bAVMs presented with hemorrhage, compared with 25 patients (51%) with bAVMs that were not associated with TN (p < 0.01). TN-associated bAVMs were overall smaller than non-TN-associated bAVMs, but the difference was not statistically significant (1.71 cm vs 2.22 cm, p = 0.117), and the Spetzler-Martin grades were similar. Six patients (75%) underwent radiosurgery to the bAVM (mean dose 1800 cGy, mean target volume 0.563 cm3) and had complete resolution of TN symptoms (100%). The mean time from radiosurgery to TN resolution was 193 (range 21-360) days, and 83.3% of treated TN-associated bAVMs were obliterated via radiosurgery. Two patients (12.5%) were recommended for conservative management, with one undergoing subsequent rhizotomies and another patient died of hemorrhage during follow-up.

CONCLUSIONS

TN-associated bAVM is a rare condition with limited evidence for management guidance. Radiosurgery can be safe and effective in achieving durable TN control in patients with TN-associated bAVMs. Despite their deep location and unruptured presentation, obliteration can reach 83.3% with radiosurgery.

摘要

目的

三叉神经痛作为脑动静脉畸形(bAVM)的首发症状较为少见。报道的病例治疗偏向手术,因为这些深部的 bAVM 很罕见。在此,作者报告了他们机构中以同侧三叉神经痛(TN)为首发症状的 bAVM 患者的治疗和结局。

方法

这是一项回顾性队列研究。作者机构的 bAVM 数据库中检索了桥脑、脑池、脑干、三叉神经或天幕位置的非遗传性出血性毛细血管扩张症 bAVM。对有完整数据的患者进行搜索,以寻找三叉神经痛或“面部疼痛”作为首发症状,且 TN 与 bAVM 同侧。分析患者的人口统计学、TN 和 bAVM 特征、管理策略以及 bAVM 和 TN 管理的结果。

结果

发现 57 例桥旁 bAVM,其中 8 例(14.0%)因同侧 TN 而被发现,包括 4 例(50%)患者出现 V2 分布的面部疼痛。5 例患者(62.5%)初始接受卡马西平治疗,2 例(25%)接受多次神经根切断术,1 例(12.5%)接受微血管减压术。与无 TN 相关的 bAVM 患者相比,有 8 例(100%)无 TN 相关的 bAVM 患者发生出血(8 例[100%])(p < 0.01)。与无 TN 相关的 bAVM 相比,TN 相关的 bAVM 总体较小,但差异无统计学意义(1.71 cm 比 2.22 cm,p = 0.117),Spetzler-Martin 分级相似。6 例患者(75%)接受 bAVM 放射外科治疗(平均剂量 1800 cGy,平均靶体积 0.563 cm3),TN 症状完全缓解(100%)。放射外科治疗至 TN 缓解的平均时间为 193(21-360)天,83.3%的治疗性 TN 相关 bAVM 经放射外科治疗闭塞。2 例患者(12.5%)建议保守治疗,其中 1 例随后行神经根切断术,另 1 例患者在随访期间死于出血。

结论

TN 相关的 bAVM 是一种罕见的疾病,治疗指导有限。放射外科治疗可以安全有效地控制 TN 相关 bAVM 患者的 TN 症状。尽管位置较深且未破裂,但放射外科治疗的闭塞率仍可达 83.3%。

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