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中国循证儿科杂志

中国循证儿科杂志 ›› 2022, Vol. 17 ›› Issue (5): 363-367.DOI: 10.3969/j.issn.1673-5501.2022.05.007

• 论著 • 上一篇    下一篇

迷走神经刺激治疗婴幼儿药物难治性癫的病例系列报告

马嘉翼1,谢涵1,刘庆祝2,王若凡2,季涛云1,2,蔡立新2,姜玉武1，2,吴晔1，2   

  1. 北京大学第一医院北京，100034;1儿科，2儿童癫中心
  • 收稿日期:2022-05-15 修回日期:2022-06-19 出版日期:2022-10-25 发布日期:2022-10-25
  • 通讯作者: 吴晔

Vagus nerve stimulation in the treatment for infants with drug-resistant epilepsy: A case series report

MA Jiayi1, XIE Han1, LIU Qingzhu2, WANG Ruofan2, JI Taoyun1,2, CAI Lixin2, JIANG Yuwu1,2, WU Ye1,2   

  1. Peking University First Hospital, Beijing 100034,China;1 Department of Pediatrics, 2 Children Epilepsy Center
  • Received:2022-05-15 Revised:2022-06-19 Online:2022-10-25 Published:2022-10-25
  • Contact: WU Ye,email:dryewu@263.net

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摘要： 背景：迷走神经刺激（VNS）治疗儿童药物难治性癫（DRE）国内年龄适应证为3岁以上，目前临床在超年龄适应证应用VNS治疗DRE婴幼儿，但其有效性和安全性研究很少。 目的：评估VNS治疗DRE婴幼儿术后的长期疗效、安全性及疗效相关因素。 设计：病例系列报告。 方法：纳入2015年3月至2021年3月于北京大学第一医院癫中心行VNS的DRE婴幼儿，且VNS术后规律程控随访至少1年、程控期间刺激电流参数＞1 mA。排除VNS期间接受新增抗癫发作治疗（癫外科手术治疗、生酮饮食、应用糖皮质激素或促肾上腺皮质激素），因各种原因而导致VNS程控关机、中断的患儿。以患儿家长癫日记所记录的所有发作类型的频率作为主要评估依据，行随访有效率（末次随访点较VNS植入前3个月发作频率减少≥50%）和癫发作减少率、考察临床信息及程控参数对疗效和安全性的影响。 主要结局指标：VNS术后有效率和癫发作减少率。 结果：符合本文纳入排除标准的VNS治疗DRE婴幼儿25例，男16例（64%）、女9例。癫起病年龄中位数4.9月，癫病程中位数1.7年，VNS治疗前每月发作中位数555次。VNS电极植入中位年龄2.3岁。病程中曾应用抗癫发作药物（ASM）3~10种，VNS治疗前3个月应用ASM及其他治疗1~4种，能明确DRE病因12例（48%），病程中曾诊断癫综合征18例（72%），存在4、3、2和1种发作形式分别为4、5、6和10例，25例VNS治疗的DRE患儿均存在发育迟缓。VNS术后随访中位数2.4年。VNS术后1年癫发作率最高增加1 367%，最低减少100%，中位数减少52.8%，VNS术后2年癫发作率最高增加1 712%，最低减少100%，中位数减少59.5%。末次随访时有效率为64%，无发作率为20%（5/25），最长随访2年5个月无发作。VNS术后1年、2年有效率均为60%（15/25，9/15）。VNS术后1年15例有效病例中1例在VNS术后2年转为无效；VNS术后1年10例无效病例中2例在术后2年转为有效。未观察到围术期相关不良反应及设备相关不良反应，4例在程控时出现刺激相关不良反应，其中2例为一过性咳嗽，2例为咽部不适，均可在短时间内耐受。疗效相关因素分析有效组（16例）和无效组（9例）临床信息、末次程控参数和术后ASM调整情况差异均无统计学意义。 结论：DRE婴幼儿VNS治疗随访1和2年有效率均可达到60%，且安全性较好；VNS治疗DRE的年龄下限仍需进一步评估。

关键词: 迷走神经刺激, 药物难治性癫痫, 婴幼儿, 有效性

Abstract: Background：The indication of vagus nerve stimulation (VNS) is approved for children with drugresistant epilepsy (DRE) older than three years old in China. Nowadays, VNS treatment is also used offlabel to treat infants with DRE in clinical practice, but there is still a lack of relevant research on its efficacy and safety. Objective：To evaluate the longterm efficacy, safety and efficacyrelated factors of VNS treatment for infants with DRE. Design：Case series report. Methods：This study included infants with DRE after VNS treatment in the children epilepsy center of Peking University First Hospital between March 2015 and March 2021. All the patients were regularly programmed and followed up after VNS implantation for at least 1 year with current output over 1mA. Patients who received other newadded antiseizure therapy including epileptic surgery, ketogenic diet, glucocorticoid and adrenocorticotropic hormone, or turned off VNS due to any reason were excluded. The frequency of all seizure types recorded in the epilepsy diary of parents was calculated to evaluate followup responder rate defined as seizure frequency decreasing by ≥ 50% at the last followup compared with 3 months before VNS implantation，and seizure frequency reduction rate so as to investigate the effect of clinical information and programmed parameters on the efficacy and safety. Main outcome measures：Responder rate and seizure frequency reduction rate after VNS treatment. Results：Twentyfive infants with DRE after VNS treatment met the inclusion and exclusion criteria, including 16 males (64%) and 9 females. The median age of seizure onset was 4.9 months. The median duration of epilepsy was 1.7 years. The median seizure frequency was 555 times per month, and the median age at VNS implantation was 2.3 years. The number of historical antiseizure medications (ASMs) used and the number of ASMs used 3 months before VNS implantation ranged 310 and 14, respectively. Twelve cases (48%) had specific DRE etiology, and eighteen (72%) were diagnosed with the epileptic syndrome. Four, five, six and ten cases had 4, 3, 2 and 1 seizure types, respectively. All the twentyfive cases of DRE infants implanted with VNS had developmental delay. The median postoperative followup duration was 2.4 years. The change rate of seizure frequency ranged from +1367% to -100% at 1year followup, with a median change rate of -52.8%. The change rate of seizure frequency ranged from +1712% to -100% at 2year followup, with a median change rate of -59.5%. At the last followup, the responder rate was 64%, the seizurefree rate was 20% (5/25) , and the longest seizurefree duration during followup was 2 years and 5 months. The responder rate for 1 year and 2 years after VNS were both 60% (15/25, 9/25). One of fifteen responders at 1 year after VNS became a nonresponder at 2 years after VNS. Two of ten nonresponders at 1 year after VNS became responders at 2 years after VNS. No perioperative and devicerelated adverse reactions were observed, and 4 patients developed stimulationrelated adverse reactions during programming, including 2 cases of transient cough and 2 cases of pharyngeal discomfort, which could be tolerated in a short time. There were no significant differences in clinical information, parameters at the last followup and postoperative ASMs adjustment between responders (16 cases) and nonresponders (9 cases) during VNS efficacyrelated factors analysis. Conclusion：The responder rate of VNS treatment for infants with DRE could reach 60% at 1 and 2 years of followup, and the safety of VNS treatment was good. The lower age limit of VNS still needs to be further evaluated.

Key words: Vagus nerve stimulation, Drug-resistant epilepsy, Infants, Efficacy