Objective To study the clinical features of children with autoimmune encephalitis (AE) secondary to epidemic encephalitis B (EEB). Methods A retrospective analysis was performed on the medical data of five children with EEB with "bipolar course" who were treated in Children's Hospital Affiliated to Zhengzhou University from January 2020 to June 2022. Results Among the five children, there were three boys and two girls, with a median age of onset of 7 years (range 3 years 9 months to 12 years) and a median time of 32 (range 25-37) days from the onset of EEB to the appearance of AE symptoms. The main symptoms in the AE stage included dyskinesia (5/5), low-grade fever (4/5), mental and behavioral disorders (4/5), convulsion (2/5), severe disturbance of consciousness (2/5), and limb weakness (1/5). Compared with the results of cranial MRI in the acute phase of EEB, the lesions were enlarged in 3 children and unchanged in 2 children showed on cranial MRI in the AE stage. In the AE stage, four children were positive for anti-N-methyl-D-aspartate receptor antibody (one was also positive for anti-γ-aminobutyric acid type B receptor antibody), and one was negative for all AE antibodies. All five children in the AE stage responded to immunotherapy and were followed up for 3 months, among whom one almost recovered and four still had neurological dysfunction. Conclusions EEB can induce AE, with anti-N-methyl-D-aspartate receptor encephalitis as the most common disease. The symptoms in the AE stage are similar to those of classical anti-N-methyl-D-aspartate receptor encephalitis. Immunotherapy is effective for children with AE secondary to EEB, and the prognosis might be related to neurological dysfunction in the acute phase of EEB.
Key words
Autoimmune encephalitis /
Epidemic encephalitis B /
Bipolar course /
Child
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References
1 Pradhan S, Gupta RK, Singh MB, et al. Biphasic illness pattern due to early relapse in Japanese-B virus encephalitis[J]. J Neurol Sci, 2001, 183(1): 13-18. PMID: 11166788. DOI: 10.1016/s0022-510x(00)00453-6.
2 Prüss H. Postviral autoimmune encephalitis: manifestations in children and adults[J]. Curr Opin Neurol, 2017, 30(3): 327-333. PMID: 28234798. DOI: 10.1097/WCO.0000000000000445.
3 Armangue T, Spatola M, Vlagea A, et al. Frequency, symptoms, risk factors, and outcomes of autoimmune encephalitis after herpes simplex encephalitis: a prospective observational study and retrospective analysis[J]. Lancet Neurol, 2018, 17(9): 760-772. PMID: 30049614. PMCID: PMC6128696. DOI: 10.1016/S1474-4422(18)30244-8.
4 Ma J, Zhang T, Jiang L. Japanese encephalitis can trigger anti-N-methyl-D-aspartate receptor encephalitis[J]. J Neurol, 2017, 264(6): 1127-1131. PMID: 28470592. DOI: 10.1007/s00415-017-8501-4.
5 Ma J, Han W, Jiang L. Japanese encephalitis-induced anti-N-methyl-d-aspartate receptor encephalitis: a hospital-based prospective study[J]. Brain Dev, 2020, 42(2): 179-184. PMID: 31563418. DOI: 10.1016/j.braindev.2019.09.003.
6 Ma J, Jiang L. Outcome of children with Japanese encephalitis and predictors of outcome in southwestern China[J]. Trans R Soc Trop Med Hyg, 2013, 107(10): 660-665. PMID: 23900120. DOI: 10.1093/trstmh/trt064.
7 Luo H, Ding X, Li Y, et al. Clinical characteristics of children with anti-N-methyl-d-aspartate receptor encephalitis after Japanese encephalitis[J]. Pediatr Neurol, 2022, 130: 46-52. PMID: 35325660. DOI: 10.1016/j.pediatrneurol.2022.02.004.
8 迟博闻, 王佳伟. 单纯疱疹病毒感染后自身免疫性脑炎的研究进展[J]. 首都医科大学学报, 2021, 42(3): 341-346. DOI: 10.3969/j.issn.1006-7795.2021.03.001.
9 Jiang Y, Yao Z, Jiang L, et al. Virus reactivation after immunotherapy of anti-NMDAR encephalitis secondary to herpes simplex encephalitis: a case report[J]. Brain Dev, 2021, 43(10): 1057-1060. PMID: 34243963. DOI: 10.1016/j.braindev.2021.06.005.
10 Liu B, Liu J, Sun H, et al. Autoimmune encephalitis after Japanese encephalitis in children: a prospective study[J]. J Neurol Sci, 2021, 424: 117394. PMID: 33773410. DOI: 10.1016/j.jns.2021.117394.
11 Dalmau J, Graus F. Antibody-mediated encephalitis[J]. N Engl J Med, 2018, 378(9): 840-851. PMID: 29490181. DOI: 10.1056/NEJMra1708712.
12 姚远, 彭斌, 王晓慧, 等. 抗N-甲基-D-天冬氨酸受体脑炎前驱期与前驱事件[J]. 中华医学杂志, 2017, 97(19): 1469-1473. PMID: 28535637. DOI: 10.3760/cma.j.issn.0376-2491.2017.19.009.
13 Si Z, Wang A, Liu J, et al. Typical clinical and imaging manifestations of encephalitis with anti-γ-aminobutyric acid B receptor antibodies: clinical experience and a literature review[J]. Neurol Sci, 2019, 40(4): 769-777. PMID: 30671737. DOI: 10.1007/s10072-018-3679-5.
14 Joubert B, Dalmau J. The role of infections in autoimmune encephalitides[J]. Rev Neurol (Paris), 2019, 175(7-8): 420-426. PMID: 31371185. DOI: 10.1016/j.neurol.2019.07.004.
15 Shu Y, Guo J, Ma X, et al. Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is associated with IRF7, BANK1 and TBX21 polymorphisms in two populations[J]. Eur J Neurol, 2021, 28(2): 595-601. PMID: 33065758. DOI: 10.1111/ene.14596.
16 Liu J, Liu L, Kang W, et al. Cytokines/chemokines: potential biomarkers for non-paraneoplastic anti-N-methyl-D-aspartate receptor encephalitis[J]. Front Neurol, 2020, 11: 582296. PMID: 33408682. PMCID: PMC7779630. DOI: 10.3389/fneur.2020.582296.