Søgaard Anna, Poulsen Charlotte Aaberg, Belhouche Nadia Zeeberg, Thybo Alberte, Hovet Siv Tonje Faret, Larsen Lykke, Nilsson Christine, Blaabjerg Morten, Nissen Mette Scheller
Department of Clinical Research, University of Southern Denmark, 5230 Odense, Denmark.
Department of Nuclear Medicine, Odense University Hospital, 5000 Odense, Denmark.
Biomedicines. 2024 Aug 27;12(9):1953. doi: 10.3390/biomedicines12091953.
It is well known that -methyl-D-aspartate receptor encephalitis (NMDARE) can be triggered by infectious encephalitis such as herpes simplex virus 1 encephalitis (HSE). However, the incidence of post-HSE NMDARE in Denmark is unknown. We reviewed literature cases and compared these to retrospectively identified cases of post-HSE NMDARE in Denmark, using a national cohort database of autoimmune encephalitis (AE) and two regional databases of infectious encephalitis patients. We identified 80 post-HSE NMDARE cases in the literature, 66% being children, who more often presented movement disorders, decreased consciousness, and sleep disturbances compared to adults. Eight patients with post-HSE NMDARE were identified from the national cohort database of AE, none being children. Forty-four HSE patients were identified from the regional infectious encephalitis databases. Of these, 16 (36%) fulfilled the Graus criteria for probable/definite NMDARE, and eight (18%) presented a prolonged/relapsing disease course. Ten (23%) were tested for AE during hospitalization. Six (14%) had leftover cerebrospinal fluid available for retrospective autoantibody testing. One out of these six patients (17%) harbored NMDARE antibodies. Thus, in total, nine post-HSE NMDARE patients have been identified in Denmark from 2009 to 2021. Comparing the adult Danish patients to the literature, Danish patients were older, but the clinical phenotype and paraclinical findings were similar. Overall, the incidence of adult post-HSE NMDARE in the Region of Southern Denmark was 0.17 per million people per year and only 7% of adult HSE patients in the region were diagnosed with post-HSE NMDARE. Our findings suggest that adult patients are still underdiagnosed and the absence of pediatric cases diagnosed with post-HSE NMDARE in Denmark is highly concerning.
众所周知，N-甲基-D-天冬氨酸受体脑炎（NMDARE）可由单纯疱疹病毒1型脑炎（HSE）等感染性脑炎引发。然而，丹麦HSE后NMDARE的发病率尚不清楚。我们回顾了文献中的病例，并将其与丹麦通过回顾性研究确定的HSE后NMDARE病例进行比较，使用了一个全国性的自身免疫性脑炎（AE）队列数据库和两个感染性脑炎患者区域数据库。我们在文献中确定了80例HSE后NMDARE病例，其中66%为儿童，与成人相比，儿童更常出现运动障碍、意识减退和睡眠障碍。从AE全国队列数据库中确定了8例HSE后NMDARE患者，均非儿童。从区域感染性脑炎数据库中确定了44例HSE患者。其中，16例（36%）符合Graus可能/确诊NMDARE标准，8例（18%）呈现病程延长/复发。10例（23%）在住院期间接受了AE检测。6例（14%）有剩余脑脊液可用于回顾性自身抗体检测。这6例患者中有1例（17%）存在NMDARE抗体。因此，2009年至2021年期间，丹麦共确定了9例HSE后NMDARE患者。将丹麦成年患者与文献中的患者进行比较，丹麦患者年龄更大，但临床表型和辅助检查结果相似。总体而言，丹麦南部地区成年HSE后NMDARE的发病率为每年每百万人0.17例，该地区只有7%的成年HSE患者被诊断为HSE后NMDARE。我们的研究结果表明，成年患者仍未得到充分诊断，丹麦未诊断出儿童HSE后NMDARE病例的情况令人高度担忧。
