Department of Biomedicine, Neurosciences and Advanced Diagnostics, University of Palermo, Palermo, Italy.
Neurology Unit, Department of Diagnostic and Therapeutic Radiology & Stroke, AOU Policlinico, Palermo, Italy.
Neurol Sci. 2024 Apr;45(4):1429-1436. doi: 10.1007/s10072-023-07189-7. Epub 2023 Nov 27.
Myelin oligodendrocyte glycoprotein-immunoglobulin G associated disease (MOGAD) is an autoimmune demyelinating disorder of the central nervous system (CNS) which usually occurs with recurrent optic neuritis, transverse myelitis, acute disseminating encephalomyelitis, or brainstem encephalitis. To date, the anti-CD 20 drug rituximab (RTX) is employed in MOGAD although some authors reported the efficacy of Tocilizumab (TCZ) in refractory patients. We present the case of a woman affected by refractory MOGAD who was treated with TCZ after therapy with RTX had failed to prevent relapses. We also conducted a current literature review on TCZ use in MOGAD. A 57-year-old Caucasian woman affected by MOGAD with severe motor impairment and cognitive dysfunction was treated from 2020 to February 2022 with RTX. However, she experienced progressive clinical and cognitive worsening associated with white matter lesions mimicking leukodystrophy. In February 2022, the patient started therapy with TCZ administered with improvement of cognitive performance, walking ability, and brainstem functions. During TCZ, our patient reached the condition of NEDA-3 (no relapse, no increase in disability, no MRI activity on neuroimaging follow-up performed in September 2023). Moreover, the patient experienced paucisymptomatic SARS-CoV-2 infection that did not modify TCZ schedule. To date, there are few evidence on the efficacy and safety of TCZ in MOGAD. However, all the reviewed cases showed that TCZ represents an effective therapy in drug-resistant MOGAD. Our case highlights the efficacy of TCZ in drug resistant MOGAD and strengthens previous reports of TCZ safety and efficacy in MOGAD.
髓鞘少突胶质细胞糖蛋白免疫球蛋白 G 相关疾病（MOGAD）是一种中枢神经系统（CNS）的自身免疫性脱髓鞘疾病，通常伴有复发性视神经炎、横贯性脊髓炎、急性播散性脑脊髓炎或脑干脑炎。迄今为止，抗 CD20 药物利妥昔单抗（RTX）用于治疗 MOGAD，尽管一些作者报道了托珠单抗（TCZ）在难治性患者中的疗效。我们报告了一例难治性 MOGAD 患者的病例，该患者在 RTX 治疗失败后，使用 TCZ 进行治疗。我们还对 TCZ 在 MOGAD 中的应用进行了当前文献综述。一名 57 岁的白人女性患有 MOGAD，伴有严重的运动障碍和认知功能障碍，从 2020 年到 2022 年 2 月，她接受了 RTX 治疗。然而，她经历了进行性的临床和认知恶化，伴有类似于白质营养不良的白质病变。2022 年 2 月，患者开始接受 TCZ 治疗，认知表现、行走能力和脑干功能均有所改善。在 TCZ 治疗期间，我们的患者达到了 NEDA-3（无复发、无残疾加重、神经影像学随访中无 MRI 活动）的状态。此外，患者经历了症状轻微的 SARS-CoV-2 感染，但并未改变 TCZ 的治疗计划。迄今为止，关于 TCZ 在 MOGAD 中的疗效和安全性的证据较少。然而，所有回顾的病例都表明，TCZ 是治疗耐药性 MOGAD 的有效药物。我们的病例强调了 TCZ 在耐药性 MOGAD 中的疗效，并加强了之前关于 TCZ 在 MOGAD 中的安全性和疗效的报告。
