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一种治疗耐药性局灶性觉知性癫痫发作的新策略:丘脑特定核团深部脑刺激。病例说明。

A new strategy for treating drug-resistant focal aware seizures: thalamic specific nuclei deep brain stimulation. Illustrative case.

作者信息

Vilela-Filho Osvaldo, Silva-Filho Hélio F, Goulart Lissa C, Ragazzo Paulo C, Arruda Francisco M

机构信息

1Division of Neurosurgery, Department of Surgery, Medical School, Federal University of Goiás, Goiânia, Brazil.

Departments of2Neurosurgery, and.

出版信息

J Neurosurg Case Lessons. 2023 Aug 21;6(8). doi: 10.3171/CASE23303.

DOI:10.3171/CASE23303
PMID:37728299
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10555561/
Abstract

BACKGROUND

Focal aware seizures (FASs) are relatively common and frequently pharmaco-resistant. If the seizure onset zone (SOZ) is in eloquent cortical areas, making resective surgery risky and inadvisable, deep brain stimulation (DBS) of the anterior nucleus of the thalamus, which is efficacious in less than half of the cases, has been the main alternative. New targets should be searched to address this deficiency. The present study aims to determine if DBS of different thalamic specific nuclei can modulate the abnormal electrical activity of the SOZ located in their respective cortical projection areas. Herein, the authors present the first patient in an ongoing trial.

OBSERVATIONS

A 60-year-old female patient presented with 25-year history of pharmaco-resistant focal aware visual seizures frequently evolving to focal impaired awareness seizures. The SOZ was in the right occipital lobe (positron emission tomography-computed tomography/video electroencephalography). Magnetic resonance imaging was normal. She underwent ipsilateral lateral geniculate nucleus (LGN) DBS procedure. After a 24-month follow-up, seizure frequency decreased by 97%, improving quality of life and daily functioning without complications.

LESSONS

This is the first time the LGN has been targeted in humans. The results support the hypothesis that led to this study. This strategy represents a paradigm shift in the way of treating pharmaco-resistant FASs not amenable to resective surgery.

摘要

背景

局灶性觉知性癫痫(FASs)相对常见且常常耐药。如果癫痫发作起始区(SOZ)位于明确的皮质区域,使得切除性手术具有风险且不可取,丘脑前核的深部脑刺激(DBS)在不到一半的病例中有效,一直是主要的替代方法。应寻找新的靶点来解决这一不足。本研究旨在确定不同丘脑特定核团的DBS是否能调节位于其各自皮质投射区域的SOZ的异常电活动。在此,作者介绍了一项正在进行的试验中的首例患者。

观察结果

一名60岁女性患者有25年耐药性局灶性觉知性视觉癫痫病史,频繁演变为局灶性意识障碍性癫痫。SOZ位于右侧枕叶(正电子发射断层扫描 - 计算机断层扫描/视频脑电图)。磁共振成像正常。她接受了同侧外侧膝状体(LGN)DBS手术。经过24个月的随访,癫痫发作频率降低了97%,改善了生活质量和日常功能且无并发症。

经验教训

这是首次在人类中靶向LGN。结果支持了导致本研究的假设。这种策略代表了治疗不适合切除性手术的耐药性FASs方式的范式转变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/252004ccd8c9/CASE23303f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/9a80806fdcf1/CASE23303f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/8f24ee98d171/CASE23303f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/1f4a5c65ae4d/CASE23303f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/252004ccd8c9/CASE23303f4.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/9a80806fdcf1/CASE23303f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/8f24ee98d171/CASE23303f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/1f4a5c65ae4d/CASE23303f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/109a/10555561/252004ccd8c9/CASE23303f4.jpg

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本文引用的文献

1
Ventral intermediate nucleus deep brain stimulation for treatment-resistant focal aware motor seizures: illustrative case.腹侧中间核深部脑刺激治疗耐药性局灶性觉知运动性癫痫:病例说明
J Neurosurg Case Lessons. 2023 Apr 3;5(14). doi: 10.3171/CASE2320.
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Experience and consensus on stimulation of the anterior nucleus of thalamus for epilepsy.丘脑前核刺激治疗癫痫的经验和共识。
Epilepsia. 2021 Dec;62(12):2883-2898. doi: 10.1111/epi.17094. Epub 2021 Oct 26.
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Incidence and Prevalence of Drug-Resistant Epilepsy: A Systematic Review and Meta-analysis.
耐药性癫痫的发病率和患病率:系统评价和荟萃分析。
Neurology. 2021 Apr 27;96(17):805-817. doi: 10.1212/WNL.0000000000011839. Epub 2021 Mar 15.
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Stimulation of the Anterior Nucleus of the Thalamus for Epilepsy: A Canadian Experience.丘脑前核刺激治疗癫痫:加拿大的经验。
Can J Neurol Sci. 2021 Jul;48(4):469-478. doi: 10.1017/cjn.2020.230. Epub 2020 Oct 16.
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Anterior thalamic nucleus deep brain stimulation for refractory epilepsy: Preliminary results in our first 5 patients.丘脑前核电刺激治疗耐药性癫痫:我们前 5 例患者的初步结果。
Neurochirurgie. 2020 Aug;66(4):252-257. doi: 10.1016/j.neuchi.2020.03.001. Epub 2020 Jun 1.
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The Epidemiology of Epilepsy.癫痫的流行病学。
Neuroepidemiology. 2020;54(2):185-191. doi: 10.1159/000503831. Epub 2019 Dec 18.
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Safety and efficacy of adjunctive cenobamate (YKP3089) in patients with uncontrolled focal seizures: a multicentre, double-blind, randomised, placebo-controlled, dose-response trial.cenobamate(YKP3089)辅助治疗未得到控制的局灶性癫痫发作患者的安全性和有效性:一项多中心、双盲、随机、安慰剂对照、剂量反应试验。
Lancet Neurol. 2020 Jan;19(1):38-48. doi: 10.1016/S1474-4422(19)30399-0. Epub 2019 Nov 14.
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The Role of Anterior Thalamic Deep Brain Stimulation as an Alternative Therapy in Patients with Previously Failed Vagus Nerve Stimulation for Refractory Epilepsy.丘脑前深部脑刺激作为迷走神经刺激治疗难治性癫痫失败患者的替代疗法的作用
Stereotact Funct Neurosurg. 2019;97(3):176-182. doi: 10.1159/000502344. Epub 2019 Sep 18.
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Brain-responsive neurostimulation for epilepsy (RNS System).用于癫痫治疗的脑响应神经刺激(RNS系统)。
Epilepsy Res. 2019 Jul;153:68-70. doi: 10.1016/j.eplepsyres.2019.02.003. Epub 2019 Feb 20.
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Epilepsia. 2018 Feb;59(2):273-290. doi: 10.1111/epi.13964. Epub 2017 Dec 7.