MOG Antibody-Associated Disease in the Setting of Metastatic Melanoma Complicated by Immune Checkpoint Inhibitor Use.

IF 7.8 1区 医学 Q1 CLINICAL NEUROLOGY
Stephanie B Syc-Mazurek, Hannah Zhao-Fleming, Yong Guo, Nanthaya Tisavipat, John J Chen, Anastasia Zekeridou, Ioannis Kournoutas, Jacob J Orme, Matthew S Block, Claudia F Lucchinetti, Rafid Mustafa, Eoin P Flanagan
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引用次数: 0

Abstract

Objectives: Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an autoimmune demyelinating disease rarely associated with malignancy. We report the clinical, MRI, immunopathology, and treatment response in a person with MOGAD and melanoma.

Methods: This is a case report of a person with a multidisciplinary evaluation at a tertiary referral center.

Results: A 52-year-old man presented with progressive encephalomyelitis that led to identification of metastatic melanoma. Investigations revealed positive MOG-IgG at high titers in serum (1:1,000; normal, <1:20) and CSF (1:4,096; normal, <1:2). MRI demonstrated multifocal T2 lesions with enhancement in the brain and spine. Brain biopsy showed demyelination and inflammation. MOG immunostaining was not present in the tumor tissue. He initially improved with methylprednisolone, plasmapheresis, prolonged oral steroid taper, and cancer-directed treatment with BRAF and MEK 1/2 inhibitors, but then developed bilateral optic neuritis. IV immunoglobulin (IVIG) was initiated. Five months later, he developed metastases and immune checkpoint inhibitor (ICI) treatment was started, which precipitated optic neuritis and myelitis despite IVIG and prednisone. Tocilizumab, an interleukin-6 receptor blocker, was started with excellent and sustained clinical and radiologic response.

Discussion: This case revealed a presentation of MOGAD concurrent with melanoma without tumor MOG immunostaining. We highlight tocilizumab as a dual-purpose treatment of MOGAD and the neurologic immune-related adverse effect of ICI.

因使用免疫检查点抑制剂而并发转移性黑色素瘤的 MOG 抗体相关疾病。
目的:髓鞘少突胶质细胞糖蛋白抗体相关疾病(MOGAD)是一种自身免疫性脱髓鞘疾病,很少与恶性肿瘤相关。我们报告了一名 MOGAD 和黑色素瘤患者的临床、磁共振成像、免疫病理和治疗反应:这是一份病例报告,患者在一家三级转诊中心接受了多学科评估:一名52岁的男性因进行性脑脊髓炎就诊,最终被确诊为转移性黑色素瘤。检查结果显示血清中MOG-IgG高滴度阳性(1:1,000;正常):本病例显示 MOGAD 与黑色素瘤并发,但无肿瘤 MOG 免疫染色。我们强调托西珠单抗具有治疗 MOGAD 和 ICI 神经系统免疫相关不良反应的双重用途。
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来源期刊
CiteScore
15.60
自引率
2.30%
发文量
219
审稿时长
8 weeks
期刊介绍: Neurology Neuroimmunology & Neuroinflammation is an official journal of the American Academy of Neurology. Neurology: Neuroimmunology & Neuroinflammation will be the premier peer-reviewed journal in neuroimmunology and neuroinflammation. This journal publishes rigorously peer-reviewed open-access reports of original research and in-depth reviews of topics in neuroimmunology & neuroinflammation, affecting the full range of neurologic diseases including (but not limited to) Alzheimer's disease, Parkinson's disease, ALS, tauopathy, and stroke; multiple sclerosis and NMO; inflammatory peripheral nerve and muscle disease, Guillain-Barré and myasthenia gravis; nervous system infection; paraneoplastic syndromes, noninfectious encephalitides and other antibody-mediated disorders; and psychiatric and neurodevelopmental disorders. Clinical trials, instructive case reports, and small case series will also be featured.
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