Myelin oligodendrocyte glycoprotein antibody-associated disease in a patient with symptoms of aseptic meningitis who achieved spontaneous remission: A case report and review of the literature

IF 1.4 4区 医学 Q4 CLINICAL NEUROLOGY Brain & Development Pub Date : 2023-09-01 DOI:10.1016/j.braindev.2023.05.002
Naomi Hino-Fukuyo , Eiichiro Kawai , Sakiko Itoh , Shuhei Oba , Yukie Sato , Sei Abe , Yukari Ichikawa , Hiroshi Kitazawa , Yuri Atobe , Juichi Fujimori , Ichiro Nakashima , Toshiyuki Takahashi , Tetsuji Morimoto
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Abstract

Background

A few case reports have described patients with myelin oligodendrocyte glycoprotein antibody (MOG-Ab)-associated demyelinating syndrome who presented with symptoms of aseptic meningitis. All such patients required immunotherapy. We report a patient with MOG-Ab-associated disorder (MOGAD) who presented with symptoms of aseptic meningitis and improved without treatment.

Case

A 13-year-old girl presented with fever, headache, decreased appetite, and neck stiffness. Cerebrospinal fluid (CSF) analysis revealed pleocytosis and magnetic resonance imaging (MRI) showed leptomeningeal enhancement. The patient was diagnosed with aseptic meningitis at admission. However, there were no signs of recovery 4 days after admission (i.e., 8 days after disease onset). Therefore, we performed extensive investigations to identify the cause of the underlying infection and inflammation. On day 14 after admission, the serum MOG-Ab test performed at admission came back positive (1:128) and she was diagnosed with MOGAD. She was discharged on day 18 after admission, because her symptoms, CSF pleocytosis, and MRI findings had improved. About 6 weeks after discharge, MRI revealed hyperintensity without gadolinium enhancement. However, her serum MOG-Ab test was negative. We did follow-ups for 11 months but found no new neurological symptoms.

Discussion and Conclusion

To the best of our knowledge, this is the first ever report of a pediatric patient with MOGAD experiencing spontaneous remission with no demyelinating symptoms during an extended follow-up period.

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一例无菌性脑膜炎症状自行缓解患者的髓鞘少突胶质细胞糖蛋白抗体相关疾病:病例报告和文献复习
背景一些病例报告描述了髓鞘少突胶质细胞糖蛋白抗体(MOG-Ab)相关脱髓鞘综合征的患者,他们表现出无菌性脑膜炎的症状。所有这些患者都需要免疫治疗。我们报告了一名MOG-Ab相关疾病(MOGAD)患者,其出现无菌性脑膜炎症状,未经治疗病情有所好转。病例一名13岁女孩出现发烧、头痛、食欲下降和颈部僵硬。脑脊液分析显示白细胞增多,磁共振成像显示软脑膜增强。病人入院时被诊断为无菌性脑膜炎。然而,入院后4天(即发病后8天)没有恢复迹象。因此,我们进行了广泛的调查,以确定潜在感染和炎症的原因。入院后第14天,入院时进行的血清MOG-Ab检测结果呈阳性(1:128),她被诊断为MOGAD。她在入院后第18天出院,因为她的症状、脑脊液白细胞增多症和MRI检查结果有所改善。出院后约6周,MRI显示高信号,钆未增强。然而,她的血清MOG-Ab测试呈阴性。我们进行了11个月的随访,但没有发现新的神经系统症状。讨论和结论据我们所知,这是有史以来第一例儿童MOGAD患者在长期随访期间出现自发缓解,无脱髓鞘症状的报告。
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来源期刊
Brain & Development
Brain & Development 医学-临床神经学
CiteScore
3.60
自引率
0.00%
发文量
153
审稿时长
50 days
期刊介绍: Brain and Development (ISSN 0387-7604) is the Official Journal of the Japanese Society of Child Neurology, and is aimed to promote clinical child neurology and developmental neuroscience. The journal is devoted to publishing Review Articles, Full Length Original Papers, Case Reports and Letters to the Editor in the field of Child Neurology and related sciences. Proceedings of meetings, and professional announcements will be published at the Editor''s discretion. Letters concerning articles published in Brain and Development and other relevant issues are also welcome.
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