Anti‐N‐methyl‐D‐aspartate receptor encephalitis with concurrent human herpes virus‐6A deoxyribonucleic acid detection: An autopsy case
Anti‐N‐methyl‐D‐aspartate receptor encephalitis with concurrent human herpes virus‐6A deoxyribonucleic acid detection: An autopsy case
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抗N-甲基-D-天冬氨酸受体脑炎并发人疱疹病毒-6A脱氧核糖核酸检测：尸检病例
DOI：
10.1111/neup.12881
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发表时间：
2022
期刊：
影响因子：
2.3
通讯作者：
Izawa Yoshikane
中科院分区：
文献类型：
--
作者：
Shimohama Sho;Iizuka Takahiro;Takizawa Tsubasa;Watanabe Narumi;Tezuka Toshiki;Matsuda Kosuke;Yamanoi Kazuhiro;Kanazawa Naomi;Kawamura Yoshiki;Yoshikawa Tetsushi;Suzuki Tadaki;Takao Masaki;Nakahara Jin;Izawa Yoshikane
本文报告一例合并人类疱疹病毒6型(HHV-6)的抗N-甲基-D-天冬氨酸受体(NMDAR)脑炎尸检病例。一名38岁以前健康的日本男子出现全身癫痫。脑磁共振成像(MRI)的表现并不明显，但脑脊液显示细胞增多。第11天，在脑脊液中检测到HHV-6DNA，随后检测了针对NMDAR(GluN1)NR1亚单位的抗体。由于最初怀疑是HHV-6脑炎，患者接受了磷甲酸钠和更昔洛韦的治疗，但HHV-6A拷贝数从200个(第22天)增加到2000个 拷贝/毫升(第47天)，治疗无效。随着抗NMDAR脑炎典型症状的出现，我们改变了患者的治疗方法，以对抗抗NMDAR脑炎。他反复接受一线免疫治疗，GluN1抗体效价下降。由于反复感染，他没有接受二线免疫治疗；他于第310天去世。尸检没有发现系统性肿瘤。脑的显微镜检查显示，只有海马腺角(CA)3-4区有严重的神经元稀疏，并有胶质增生。在难治性抗NMDAR脑炎病例中，可能需要早期开始积极的免疫治疗，即使是HHV-6A DNA检测也是如此，因为这种同时检测在自身免疫性脑炎中的意义尚不清楚。
We report an autopsy case of anti‐N‐methyl‐D‐aspartate (NMDA) receptor (NMDAR) encephalitis with concurrent human herpes virus‐6 (HHV‐6) A deoxyribonucleic acid (DNA) detection in cerebrospinal fluid (CSF). A 38‐year‐old previously healthy Japanese man presented with a generalized seizure. Brain magnetic resonance imaging (MRI) findings were unremarkable, but CSF revealed pleocytosis. On Day 11, HHV‐6 DNA was detected in CSF, and IgG antibodies against the NR1 subunit of the NMDAR (GluN1) were subsequently detected. Since HHV‐6 encephalitis was initially suspected, the patient was treated with foscarnet and ganciclovir, but the HHV‐6A copy number increased from 200 (Day 22) to 2000 copies/mL (Day 47), and the therapy was ineffective. As typical symptoms of anti‐NMDAR encephalitis developed, we changed the patient's treatment to combat anti‐NMDAR encephalitis. He was repeatedly treated with first‐line immunotherapy, and GluN1 antibody titer decreased. He was not treated with second‐line immunotherapy because of recurrent infections; he died on Day 310. Postmortem examinations did not show systemic tumors. Microscopic examination of the brain revealed only severe neuronal rarefaction in the hippocampal cornu ammonis (CA) 3–4 areas with gliosis. Early initiation of aggressive immunotherapy may be required in a refractory case of anti‐NMDAR encephalitis, even with HHV‐6A DNA detection, because the significance of this concurrent detection in autoimmune encephalitis remains unclear.
DOI：
10.1212/nxi.0000000000000245
发表时间：
2016-08
期刊：
Neurology(R) neuroimmunology & neuroinflammation
影响因子：
--
作者：
Linnoila JJ;Binnicker MJ;Majed M;Klein CJ;McKeon A
通讯作者：
McKeon A
DOI：
10.1016/s1474-4422(12)70310-1
发表时间：
2013-02
期刊：
The Lancet. Neurology
影响因子：
--
作者：
Titulaer MJ;McCracken L;Gabilondo I;Armangué T;Glaser C;Iizuka T;Honig LS;Benseler SM;Kawachi I;Martinez-Hernandez E;Aguilar E;Gresa-Arribas N;Ryan-Florance N;Torrents A;Saiz A;Rosenfeld MR;Balice-Gordon R;Graus F;Dalmau J
通讯作者：
Dalmau J
影响因子：
11.2
作者：
Zrzavy, Tobias;Endmayr, Verena;Hoeftberger, Romana
通讯作者：
Hoeftberger, Romana