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[1例复发型视神经炎合并脑和脊髓病变及抗髓鞘少突胶质细胞糖蛋白自身抗体患者在芬戈莫德治疗后复发]

[A case of recurrent optic neuritis associated with cerebral and spinal cord lesions and autoantibodies against myelin oligodendrocyte glycoprotein relapsed after fingolimod therapy].

作者信息

Miyazaki Teiichiro, Nakajima Hideki, Motomura Masakatsu, Tanaka Keiko, Maeda Yasuhiro, Shiraishi Hirokazu, Tsujino Akira

机构信息

Department of Neurology and Strokology, Nagasaki University Hospital.

出版信息

Rinsho Shinkeigaku. 2016 Apr 28;56(4):265-9. doi: 10.5692/clinicalneurol.cn-000756. Epub 2016 Mar 24.

DOI:10.5692/clinicalneurol.cn-000756
PMID:27010093
Abstract

A previously healthy 16-year-old girl developed sudden eye pain and visual loss in her right eye. On day 7 from onset her right visual acuity had decreased to light perception, and she underwent 5 courses of intravenous methylprednisolone therapy (IVMP, 1 g/day for 3 consecutive days per week). Her eye pain and her visual acuity had improved immediately. Eleven months later, follow-up MRI revealed three T2-hyperintense plaques involving subcortical white matter in the left occipital lobe, right frontal lobe, right thalamus, and thoracic spinal cord. We suspected the diagnosis as multiple sclerosis and treated with fingolimod. She developed recurrent optic neuritis (ON) on day 19 from fingolimod therapy, and we stopped fingolimod. For two years from onset she was admitted five times due to recurrences of ON and appearance of white matter lesion and myelitis. At 22 months, anti-myelin oligodendrocyte glycoprotein (MOG) antibodies revealed to be positive in her sera from the onset to the present. Our case report suggests that fingolimod might not be effective in anti-MOG antibody-related disorders together with anti-aquaporin-4 (AQP4) antibody-positive group.

摘要

一名既往健康的16岁女孩突然出现右眼疼痛和视力丧失。发病第7天,她的右眼视力降至光感,接受了5个疗程的静脉注射甲泼尼龙治疗(IVMP,每周连续3天,每天1 g)。她的眼痛和视力立即得到改善。11个月后,随访MRI显示左侧枕叶、右侧额叶、右侧丘脑和胸段脊髓的皮质下白质有3个T2高信号斑块。我们怀疑诊断为多发性硬化症,并使用芬戈莫德进行治疗。在开始使用芬戈莫德治疗的第19天,她出现了复发性视神经炎(ON),我们停用了芬戈莫德。从发病开始的两年里,她因ON复发、白质病变和脊髓炎出现而住院5次。在22个月时,抗髓鞘少突胶质细胞糖蛋白(MOG)抗体显示从发病到目前她的血清均为阳性。我们的病例报告表明,芬戈莫德可能对与抗MOG抗体相关的疾病以及抗水通道蛋白4(AQP4)抗体阳性组无效。

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引用本文的文献

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Cureus. 2019 Oct 30;11(10):e6040. doi: 10.7759/cureus.6040.
2
[MOG encephalomyelitis: international recommendations on diagnosis and antibody testing].[MOG 脑脊髓炎:诊断和抗体检测的国际建议]
Nervenarzt. 2018 Dec;89(12):1388-1399. doi: 10.1007/s00115-018-0607-0.
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CSF cytokine profile in MOG-IgG+ neurological disease is similar to AQP4-IgG+ NMOSD but distinct from MS: a cross-sectional study and potential therapeutic implications.
CSF 细胞因子谱在 MO-G 免疫球蛋白 G 阳性神经疾病中的表现类似于水通道蛋白 4 免疫球蛋白 G 阳性 NMOSD,但与 MS 不同:一项横断面研究及潜在的治疗意义。
J Neurol Neurosurg Psychiatry. 2018 Sep;89(9):927-936. doi: 10.1136/jnnp-2018-317969. Epub 2018 Jun 6.
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MOG encephalomyelitis: international recommendations on diagnosis and antibody testing.MOG 脑炎:诊断和抗体检测的国际建议。
J Neuroinflammation. 2018 May 3;15(1):134. doi: 10.1186/s12974-018-1144-2.