Knudtzen Fredrikke Christie, Eikeland Randi, Bremell Daniel, Quist-Paulsen Else, Johansen Isik Somuncu, Solheim Anne-Marit, Skarphédinsson Sigurdur
Clinical Center for Emerging and Vector-borne Infections, Odense University Hospital, Odense, Denmark; Department of Infectious Diseases, Odense University Hospital, Odense, Denmark; Research Unit of Infectious Diseases, Faculty of Health Sciences, University of Southern Denmark, Odense, Denmark.
The Norwegian National Advisory Unit on Tick-Borne Diseases, Sørlandet Hospital, Arendal, Norway; Faculty of Health & Sport Sciences, University of Agder, Norway.
Clin Microbiol Infect. 2022 May;28(5):649-656. doi: 10.1016/j.cmi.2021.11.001. Epub 2021 Nov 10.
Lyme neuroborreliosis (LNB) presenting with encephalitis is rare and scarcely described.
To describe the available literature on LNB encephalitis and to characterize this patient group through a Scandinavian retrospective cohort study.
Medline, Embase, Scopus, Cochrane library.
There was no discrimination on study type, time of publication or language.
Review: All articles with definite LNB and confirmed/possible encephalitis.
LNB cohorts from Denmark, Sweden and Norway 1990-2019 were screened for patients with encephalitis.
Review: Adhering to PRISMA guidelines; two authors extracted reviews and assessed quality of studies.
Data on demography, symptoms, cerebrospinal fluid findings, differential diagnostic examinations, treatment, residual symptoms, 1-year mortality were registered.
Review: 2330 articles screened on title/abstract, 281 full texts, yielding 42 articles (case reports/series or cohort studies), including 45 patients from 18 countries spanning 35 years. Altered mental status ranged from personality changes and confusion to unconsciousness. Common focal symptoms were hemiparesis, ataxia and dysarthria; seven patients had seizures. Median time from symptom onset to hospital was 2 weeks (IQR 2-90 days). Of 38 patients with available follow-up after median 12 months (IQR 5-13), 32 had fully or partially recovered, two had died.
Thirty-five patients (median age 67 years, IQR 48-76) were included. The encephalitis prevalence was 3.3% (95% CI 2.2-4.4%) among 1019 screened LNB patients. Frequent encephalitis symptoms were confusion, personality changes, aphasia, ataxia. EEGs and neuroimaging showed encephalitis in 93.8% and 20.6%, respectively. Median delay from symptom onset to hospital was 14 days (IQR 7-34), with further 7 days (IQR 3-34) delay until targeted therapy. At follow-up (median 298 days post-treatment; IQR 113-389), 65.6% had residual symptoms. None had died.
This study shows that encephalitis is an uncommon, but likely overlooked clinical manifestation of LNB. As the high frequency of residual symptoms may be related to prolonged treatment delay, prompt LNB testing of patients with encephalitis in Borrelia burgdorferi-endemic areas should be considered.
以脑炎形式出现的莱姆病神经螺旋体病（LNB）较为罕见，鲜有相关描述。
描述关于LNB脑炎的现有文献，并通过一项斯堪的纳维亚回顾性队列研究对该患者群体进行特征分析。
医学期刊数据库（Medline）、荷兰医学文摘数据库（Embase）、斯高帕斯数据库（Scopus）、考克兰图书馆。
对研究类型、发表时间或语言无限制。
综述：所有明确诊断为LNB且确诊/可能患有脑炎的文章。
对1990年至2019年丹麦、瑞典和挪威的LNB队列进行筛查，以确定患有脑炎的患者。
综述：遵循系统评价和荟萃分析优先报告的项目（PRISMA）指南；两名作者提取综述并评估研究质量。
记录人口统计学、症状、脑脊液检查结果、鉴别诊断检查、治疗、残留症状、1年死亡率等数据。
综述：通过标题/摘要筛选出2330篇文章，281篇全文，最终纳入42篇文章（病例报告/系列或队列研究），包括来自18个国家的45例患者，时间跨度为35年。精神状态改变范围从人格改变、意识模糊到昏迷。常见的局灶性症状有偏瘫、共济失调和构音障碍；7例患者有癫痫发作。从症状出现到入院的中位时间为2周（四分位间距2 - 90天）。在38例有随访数据的患者中，中位随访时间为12个月（四分位间距5 - 13），32例患者完全或部分康复，2例死亡。
纳入35例患者（中位年龄67岁，四分位间距48 - 76）。在1019例接受筛查的LNB患者中，脑炎患病率为3.3%（95%置信区间2.2 - 4.4%）。常见的脑炎症状有意识模糊、人格改变、失语、共济失调。脑电图和神经影像学检查分别显示93.8%和20.6%的患者存在脑炎表现。从症状出现到入院的中位延迟时间为14天（四分位间距7 - 34），直到开始针对性治疗又延迟了7天（四分位间距3 - 34）。在随访时（治疗后中位时间298天；四分位间距113 - 389），65.6%的患者有残留症状。无患者死亡。
本研究表明，脑炎是LNB一种不常见但可能被忽视的临床表现。由于残留症状的高发生率可能与治疗延迟时间延长有关，对于莱姆病螺旋体流行地区患有脑炎的患者，应考虑及时进行LNB检测。
