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迷走神经刺激在孕早期新发难治性癫痫持续状态中的安全有效植入及应用:一例报告

Safe and effective implantation and use of vagal nerve stimulation in new-onset refractory status epilepticus in early pregnancy: a case report.

作者信息

Jindal Malaika, Delaj Laura, Winston Joel, Goel Rishu, Bhatti Sadia, Angelova-Chee Milena, Selway Richard, Mantoan Ritter Laura

机构信息

Faculty of Life Sciences and Medicine, King's College London, London, United Kingdom.

Department of Neurology, East Kent Hospitals University National Health Service (NHS) Foundation Trust, Kent, United Kingdom.

出版信息

Front Neurol. 2023 May 15;14:1183080. doi: 10.3389/fneur.2023.1183080. eCollection 2023.

Abstract

INTRODUCTION

The management of new-onset refractory status epilepticus (NORSE) in pregnancy may be complicated by anti-seizure medication (ASM) polytherapy-associated teratogenicity. We aim to demonstrate the safety and efficacy of vagal nerve stimulation (VNS) in a pregnant patient presenting with NORSE.

CASE DESCRIPTION

A 30-year old female, at 5-weeks' gestation presented with drug-refractory myoclonic status epilepticus, responsive only to high levels of anesthetic agents. The severity of seizures did not allow extubation, and the patient remained ventilated and sedated. VNS was implanted 26 days after seizure onset. The immediate post-operative output was 0.25 mA, which was rapidly titrated up to 0.5 mA the next morning, and to 0.75 mA that afternoon. This was further increased to 1.0 mA on 3rd day post-operation, and to 1.25 mA 7 days post-op. Myoclonic jerks diminished significantly 7 days post-op, allowing extubation. Twenty days after VNS implantation, no myoclonic jerks were observed. There was also a notable neurological improvement including increased alertness and mobility, and ability to obey commands. Drug overdose was subsequently found to be the most likely etiology of her NORSE. An early pregnancy assessment 17 days after VNS implantation showed a normally sited pregnancy, normal fetal heart activity and crown-rump length. The patient remained seizure free, gained functional independence and delivered a premature but otherwise healthy baby at 33 weeks' gestation.

CONCLUSION

NORSE is challenging to manage, further compounded in pregnancy due to the teratogenicity of ASMs and ASM polytherapy. This is the first case-study to report the safe implantation and use of VNS during the first trimester of pregnancy for the management of NORSE.

摘要

引言

妊娠期间新发难治性癫痫持续状态(NORSE)的管理可能因抗癫痫药物(ASM)联合治疗相关的致畸性而变得复杂。我们旨在证明迷走神经刺激(VNS)在一名患有NORSE的孕妇中的安全性和有效性。

病例描述

一名30岁女性,妊娠5周时出现药物难治性肌阵挛性癫痫持续状态,仅对高剂量麻醉剂有反应。癫痫发作的严重程度不允许拔管,患者仍需通气和镇静。癫痫发作26天后植入VNS。术后即刻输出为0.25毫安,第二天早上迅速滴定至0.5毫安,当天下午滴定至0.75毫安。术后第3天进一步增加至1.0毫安,术后7天增加至1.25毫安。术后7天肌阵挛性抽搐明显减少,允许拔管。VNS植入20天后,未观察到肌阵挛性抽搐。还出现了明显的神经功能改善,包括警觉性和活动能力增加以及能够听从指令。随后发现药物过量是她NORSE最可能的病因。VNS植入17天后的早期妊娠评估显示妊娠位置正常、胎儿心脏活动正常且头臀长度正常。患者一直无癫痫发作,获得了功能独立性,并在妊娠33周时产下一名早产但健康的婴儿。

结论

NORSE的管理具有挑战性,由于ASM的致畸性和ASM联合治疗,在妊娠期间情况更加复杂。这是第一例报告在妊娠早期安全植入和使用VNS治疗NORSE的病例研究。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9380/10225632/a0f53af114d1/fneur-14-1183080-g0001.jpg

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