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因病灶周围炎症导致术后长期瘫痪的脑海绵状血管畸形:病例报告

Cerebral cavernous malformation with prolonged postoperative paralysis due to perilesional inflammation: illustrative case.

作者信息

Inai Soichi, Sano Noritaka, Takeuchi Yasuhide, Makino Yasuhide, Yamamoto Hattori Etsuko, Takada Shigeki, Tanji Masahiro, Mineharu Yohei, Arakawa Yoshiki

机构信息

Department of Neurosurgery, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Department of Diagnostic Pathology, Kyoto University Hospital, Kyoto, Japan.

出版信息

J Neurosurg Case Lessons. 2024 Dec 2;8(23). doi: 10.3171/CASE24570.

DOI:10.3171/CASE24570
PMID:39622022
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11616148/
Abstract

BACKGROUND

Postoperative symptom exacerbation after resection of cerebral cavernous malformations (CCMs) is usually due to surgical damage to the eloquent areas or venous outflow obstruction from injury to a developmental venous anomaly (DVA).

OBSERVATIONS

A 21-year-old right-handed female presented with headache, right limb weakness, and aphasia. Magnetic resonance imaging (MRI) revealed a 3.5-cm CCM with significant perilesional edema in the middle frontal gyrus. Despite medical treatment, her weakness worsened, necessitating emergency resection. Imaging revealed no DVA or venous obstructions. Histopathological examination revealed marked neutrophil infiltration, indicating noninfectious inflammation. One week postoperatively, MRI revealed increased edema around the resection site. Although the aphasia improved, paralysis (manual muscle testing grade 3) persisted, prompting betamethasone administration. The symptoms rapidly improved over 10 days, and the patient was discharged symptom free on day 20 with no recurrence thereafter.

LESSONS

Patients with prolonged postoperative deficits after CCM resection can experience noninfectious inflammation. Anti-inflammatory treatments such as corticosteroids may be necessary in similar cases with poor recovery from edema and symptoms. https://thejns.org/doi/10.3171/CASE24570.

摘要

背景

脑海绵状血管畸形(CCM)切除术后症状加重通常是由于手术损伤了功能区或因发育性静脉异常(DVA)损伤导致静脉流出道梗阻。

观察结果

一名21岁右利手女性,出现头痛、右肢无力和失语症状。磁共振成像(MRI)显示额叶中回有一个3.5厘米的CCM,病灶周围有明显水肿。尽管进行了药物治疗,但其无力症状仍加重,因此需要紧急手术切除。影像学检查未发现DVA或静脉梗阻。组织病理学检查显示有明显的中性粒细胞浸润,提示为非感染性炎症。术后一周,MRI显示切除部位周围水肿加重。虽然失语症状有所改善,但瘫痪症状(徒手肌力测试3级)仍然存在,遂给予倍他米松治疗。症状在10天内迅速改善,患者在第20天无症状出院,此后未复发。

经验教训

CCM切除术后出现长期功能缺损的患者可能会发生非感染性炎症。在类似因水肿和症状恢复不佳的病例中,可能需要使用皮质类固醇等抗炎治疗。https://thejns.org/doi/10.3171/CASE24570

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/77c2cf7d7d77/CASE24570_figure_3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/23242538a59a/CASE24570_figure_1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/faad32bab0cd/CASE24570_figure_2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/77c2cf7d7d77/CASE24570_figure_3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/23242538a59a/CASE24570_figure_1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/faad32bab0cd/CASE24570_figure_2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8bcd/11616148/77c2cf7d7d77/CASE24570_figure_3.jpg

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本文引用的文献

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Hyperintense Perilesional Edema on T1-Weighted Imaging in Cavernoma: A Case Report.海绵状血管瘤T1加权成像上的高信号周围水肿:一例报告
Cureus. 2024 Jan 1;16(1):e51454. doi: 10.7759/cureus.51454. eCollection 2024 Jan.
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Inflammatory Mechanisms in a Neurovascular Disease: Cerebral Cavernous Malformation.一种神经血管疾病中的炎症机制:脑海绵状血管畸形
Brain Sci. 2023 Sep 17;13(9):1336. doi: 10.3390/brainsci13091336.
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Neuroinflammation Plays a Critical Role in Cerebral Cavernous Malformation Disease.神经炎症在脑动静脉畸形疾病中发挥着关键作用。
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Cerebral Cavernous Malformation: Immune and Inflammatory Perspectives.颅内海绵状血管畸形:免疫与炎症的观点。
Front Immunol. 2022 Jun 30;13:922281. doi: 10.3389/fimmu.2022.922281. eCollection 2022.
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Inflammation and neutrophil extracellular traps in cerebral cavernous malformation.脑动静脉畸形中的炎症和中性粒细胞胞外陷阱。
Cell Mol Life Sci. 2022 Mar 25;79(4):206. doi: 10.1007/s00018-022-04224-2.
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Use of Oxidized Regenerated Cellulose as a Hemostatic Agent in Neurosurgery: Appraisals and Recommendations to Prevent Postoperative Complications and Facilitate Follow-Up.氧化再生纤维素在神经外科中的应用:作为止血剂的评估和建议,以预防术后并发症并促进随访。
Surg Technol Int. 2021 May 20;38:481-485. doi: 10.52198/21.STI.38.NS1397.
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Outcomes of Surgery for Brainstem Cavernous Malformations: A Systematic Review.脑干海绵状血管畸形手术治疗的结果:系统评价。
Stroke. 2019 Oct;50(10):2964-2966. doi: 10.1161/STROKEAHA.119.026120. Epub 2019 Sep 12.
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Intraaxial and Extraaxial Cavernous Malformation with Venous Linkage: Immune Cellular Inflammation Associated with Aggressiveness.伴有静脉交通的颅内和颅外海绵状血管畸形:与侵袭性相关的免疫细胞炎症。
World Neurosurg. 2019 Nov;131:87-89. doi: 10.1016/j.wneu.2019.07.145. Epub 2019 Jul 26.
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Synopsis of Guidelines for the Clinical Management of Cerebral Cavernous Malformations: Consensus Recommendations Based on Systematic Literature Review by the Angioma Alliance Scientific Advisory Board Clinical Experts Panel.脑海绵状血管畸形临床管理指南概要:基于血管瘤联盟科学咨询委员会临床专家小组系统文献综述的共识建议
Neurosurgery. 2017 May 1;80(5):665-680. doi: 10.1093/neuros/nyx091.
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