{"title":"Initiation of tocilizumab within 24 hours in acute necrotizing encephalopathy is related to a good outcome: a systematic review.","authors":"Yalin Ju, Chang Geng, Hongzhi Guan","doi":"10.1007/s00415-025-13355-y","DOIUrl":null,"url":null,"abstract":"<p><strong>Objective: </strong>To evaluate the clinical efficacy of tocilizumab, a interleukin-6 (IL-6) receptor blocker, for the treatment of acute necrotizing encephalopathy (ANE).</p><p><strong>Methods: </strong>PubMed, Cochrane Library, Embase, and Web of Science were searched for systematic review based on PRISMA guidelines. ANE patients treated with and without tocilizumab were included. Methodological quality was assessed independently by two authors. Data on clinical features, neuroimaging patterns and outcomes were analyzed.</p><p><strong>Results: </strong>In total, 77 cases from 21 studies were included. Most patients had fever and seizures. Respiratory viruses were common precipitating infection. All patients had bilateral thalamic lesions, and brainstem lesions were more frequently observed in the tocilizumab group (88.0% vs 64.0%, p = 0.04). The length of hospital stay was significantly longer in the tocilizumab group both before (p = 0.01) and after propensity score matching (PSM) (p = 0.02), while the mortality rate was significantly lower in tocilizumab compared with non-tocilizumab group (8.0% vs 33.3%, p = 0.02). Initiation of tocilizumab within 24 h was related to a good outcome both before (p < 0.01) and after PSM (p < 0.01). In multivariate regression analysis, elder age and < 24 h immunotherapy were independently related with good outcome (age: aOR = 1.35; 95% CI 1.06-1.74, p = 0.02; < 24 h immunotherapy: aOR = 8.60; 95% CI 1.08-68.63, p = 0.04). Brainstem lesions was an independent risk factor of poor outcome (aOR = 0.03; 95% CI 0.003-0.29, p < 0.01). Tocilizumab use was independently associated with reduced mortality (aOR = 7.95, 95%CI 1.47-43.03, p = 0.02). No adverse effects were reported.</p><p><strong>Conclusion: </strong>This review suggests that timely initiation of tocilizumab therapy within 24 h is safe, and may be an effective treatment in ANE, providing a strong rationale for a clinical trial.</p>","PeriodicalId":16558,"journal":{"name":"Journal of Neurology","volume":"272 9","pages":"618"},"PeriodicalIF":4.6000,"publicationDate":"2025-09-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Neurology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00415-025-13355-y","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Objective: To evaluate the clinical efficacy of tocilizumab, a interleukin-6 (IL-6) receptor blocker, for the treatment of acute necrotizing encephalopathy (ANE).
Methods: PubMed, Cochrane Library, Embase, and Web of Science were searched for systematic review based on PRISMA guidelines. ANE patients treated with and without tocilizumab were included. Methodological quality was assessed independently by two authors. Data on clinical features, neuroimaging patterns and outcomes were analyzed.
Results: In total, 77 cases from 21 studies were included. Most patients had fever and seizures. Respiratory viruses were common precipitating infection. All patients had bilateral thalamic lesions, and brainstem lesions were more frequently observed in the tocilizumab group (88.0% vs 64.0%, p = 0.04). The length of hospital stay was significantly longer in the tocilizumab group both before (p = 0.01) and after propensity score matching (PSM) (p = 0.02), while the mortality rate was significantly lower in tocilizumab compared with non-tocilizumab group (8.0% vs 33.3%, p = 0.02). Initiation of tocilizumab within 24 h was related to a good outcome both before (p < 0.01) and after PSM (p < 0.01). In multivariate regression analysis, elder age and < 24 h immunotherapy were independently related with good outcome (age: aOR = 1.35; 95% CI 1.06-1.74, p = 0.02; < 24 h immunotherapy: aOR = 8.60; 95% CI 1.08-68.63, p = 0.04). Brainstem lesions was an independent risk factor of poor outcome (aOR = 0.03; 95% CI 0.003-0.29, p < 0.01). Tocilizumab use was independently associated with reduced mortality (aOR = 7.95, 95%CI 1.47-43.03, p = 0.02). No adverse effects were reported.
Conclusion: This review suggests that timely initiation of tocilizumab therapy within 24 h is safe, and may be an effective treatment in ANE, providing a strong rationale for a clinical trial.
目的：评价白细胞介素-6 （IL-6）受体阻滞剂托珠单抗治疗急性坏死性脑病（ANE）的临床疗效。方法：基于PRISMA指南检索PubMed、Cochrane Library、Embase和Web of Science进行系统评价。纳入了接受和不接受托珠单抗治疗的ANE患者。方法学质量由两位作者独立评估。分析临床特征、神经影像学模式和预后数据。结果：21项研究共纳入77例。大多数患者有发烧和癫痫。呼吸道病毒是常见的诱发性感染。所有患者均有双侧丘脑病变，托珠单抗组脑干病变发生率更高（88.0% vs 64.0%, p = 0.04）。托珠单抗组在倾向评分匹配（PSM）前（p = 0.01）和后（p = 0.02）的住院时间明显更长，而托珠单抗组的死亡率明显低于非托珠单抗组（8.0% vs 33.3%, p = 0.02）。结论：本综述提示，在24小时内及时开始tocilizumab治疗是安全的，并且可能是ANE的有效治疗方法，为临床试验提供了强有力的依据。
期刊介绍：
The Journal of Neurology is an international peer-reviewed journal which provides a source for publishing original communications and reviews on clinical neurology covering the whole field.
In addition, Letters to the Editors serve as a forum for clinical cases and the exchange of ideas which highlight important new findings. A section on Neurological progress serves to summarise the major findings in certain fields of neurology. Commentaries on new developments in clinical neuroscience, which may be commissioned or submitted, are published as editorials.
Every neurologist interested in the current diagnosis and treatment of neurological disorders needs access to the information contained in this valuable journal.
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