Dorcet Guillaume, Benaiteau Marie, Bost Chloé, Mengelle Catherine, Bonneville Fabrice, Martin-Blondel Guillaume, Pariente Jérémie
Département de Neurologie, Hôpital Pierre Paul Riquet, CHU de Toulouse, Toulouse, France.
Laboratoire d'Immunologie, Institut Fédératif de Biologie, CHU de Toulouse, Toulouse, France.
Front Neurol. 2020 Feb 18;11:38. doi: 10.3389/fneur.2020.00038. eCollection 2020.
Encephalitis due to herpes simplex virus 1 (HSV-1) was described as a potential trigger for the development of anti-N-methyl-D-aspartate receptor (NMDAr) auto-immune encephalitis (AIE) within a few days to a few weeks after the infection. We assessed clinical, radiological, and biological diagnoses process, treatment response, and evolution. We report here cases of a 71-year-old man and a 57-year-old woman presenting anti-NMDAr AIE, respectively, 12 and 7 months after HSV-1 encephalitis. In both cases, the onset was brisk, and the symptoms were mainly neuropsychiatric (paranoid delirium, Capgras, and Cotard syndromes) and cognitive, with anterograde amnesia. Relapse of HSV encephalitis, epilepsy, and paraneoplastic neurologic syndromes were excluded. The clinical response to first-line treatments composed of intravenous immunoglobulins and high-dose corticosteroids was poor, whereas significant improvement was noticed after rituximab induction. Post-herpetic anti-NMDAr AIE could arise several months after infection. Clinicians must be aware of this possibility, particularly if cognitive and/or psychiatric symptoms occurred after a remitting period. In our two cases, only rituximab was associated with clinical improvement.
1型单纯疱疹病毒（HSV-1）所致脑炎被描述为在感染后数天至数周内，可能引发抗N-甲基-D-天冬氨酸受体（NMDAr）自身免疫性脑炎（AIE）的诱因。我们评估了临床、影像学和生物学诊断过程、治疗反应及病情演变。我们在此报告1例71岁男性和1例57岁女性病例，分别在HSV-1脑炎后12个月和7个月出现抗NMDAr AIE。两例起病均急，症状主要为神经精神症状（偏执性谵妄、卡普格拉综合征和科塔尔综合征）及认知症状，伴有顺行性遗忘。排除了HSV脑炎复发、癫痫及副肿瘤性神经综合征。由静脉注射免疫球蛋白和大剂量皮质类固醇组成的一线治疗临床反应不佳，而利妥昔单抗诱导治疗后有显著改善。感染后数月可能出现疱疹后抗NMDAr AIE。临床医生必须意识到这种可能性，尤其是在缓解期后出现认知和/或精神症状时。在我们的两例病例中，只有利妥昔单抗与临床改善相关。
