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病例报告:艾加莫德治疗难治性抗GQ1b抗体综合征合并重症肌无力的疗效

Case Report: Therapeutic effect of efgartigimod in refractory anti-GQ1b antibody syndrome coexisting with myasthenia gravis.

作者信息

Watanabe Keiko, Takahashi Seiya, Kanda Akane, Watanabe Takuya, Kakinuma Yuki, Yano Satoshi, Kinno Ryuta

机构信息

Department of Internal Medicine, Showa Medical University Northern Yokohama Hospital, Yokohama, Japan.

Department of Neurology, Showa Medical University Fujigaoka Hospital, Yokohama, Japan.

出版信息

Front Immunol. 2025 Jun 10;16:1605985. doi: 10.3389/fimmu.2025.1605985. eCollection 2025.

Abstract

Anti-GQ1b antibody syndrome is a spectrum of autoimmune neurological disorders that includes Miller Fisher syndrome, Guillain-Barré syndrome (GBS) with ophthalmoplegia, Bickerstaff brainstem encephalitis, and acute ophthalmoplegia without ataxia. These conditions are characterized by the presence of immunoglobulin G (IgG) antibodies targeting GQ1b gangliosides. The coexistence of anti-GQ1b antibody syndrome and myasthenia gravis (MG) is rare and presents diagnostic and therapeutic challenges. We report the case of an 84-year-old Japanese man with overlapping features of both disorders, describing his clinical course and response to add-on treatment with the neonatal Fc receptor antagonist efgartigimod. He presented with fever and diarrhea, followed by acute limb weakness. He was initially suspected of having had a stroke but was later diagnosed with GBS based on areflexia, anti-ganglioside antibody positivity, and nerve conduction abnormalities. Intravenous immunoglobulin therapy was initiated but his condition worsened, leading to respiratory failure and mechanical ventilation. Subsequently, bilateral ptosis and eye movement dysfunction emerged, prompting the consideration of MG. Anti-acetylcholine receptor antibodies and tensilon test results were positive and high-dose methylprednisolone was administered, resulting in partial improvement. Plasmapheresis was performed, but profound limb weakness and respiratory failure persisted; intravenous efgartigimod was thus introduced. Remarkably, the patient's respiratory function improved within 7 days, leading to ventilator weaning, and his limb weakness showed notable recovery. After a second cycle of efgartigimod, the patient regained speech and independent mobility, allowing transfer to a rehabilitation facility. His case underscores the diagnostic complexity of overlapping anti-GQ1b antibody syndrome and MG, and it highlights the therapeutic potential of efgartigimod in treating refractory cases of overlapping autoimmune neuromuscular syndromes. Given the rapid efficacy of efgartigimod for improving both respiratory and motor functions in this case, it is apparent that efgartigimod can be a valuable therapeutic option in complex neuromuscular autoimmune conditions.

摘要

抗GQ1b抗体综合征是一系列自身免疫性神经疾病,包括米勒·费希尔综合征、伴有眼肌麻痹的吉兰-巴雷综合征(GBS)、比克斯特法夫脑干脑炎以及无共济失调的急性眼肌麻痹。这些病症的特征是存在靶向GQ1b神经节苷脂的免疫球蛋白G(IgG)抗体。抗GQ1b抗体综合征与重症肌无力(MG)并存的情况罕见,会带来诊断和治疗方面的挑战。我们报告了一名84岁日本男性同时患有这两种疾病重叠特征的病例,描述了他的临床病程以及对添加新生儿Fc受体拮抗剂艾加莫德治疗的反应。他起初出现发热和腹泻,随后出现急性肢体无力。他最初被怀疑中风,但后来根据腱反射消失、抗神经节苷脂抗体阳性和神经传导异常被诊断为GBS。开始进行静脉注射免疫球蛋白治疗,但他的病情恶化,导致呼吸衰竭并需要机械通气。随后,出现双侧上睑下垂和眼球运动功能障碍,促使考虑MG。抗乙酰胆碱受体抗体和腾喜龙试验结果呈阳性,于是给予大剂量甲基泼尼松龙,病情有部分改善。进行了血浆置换,但严重的肢体无力和呼吸衰竭仍然存在;因此开始静脉注射艾加莫德。值得注意的是,患者的呼吸功能在7天内得到改善,得以脱机,肢体无力也有明显恢复。在第二个艾加莫德疗程后,患者恢复了言语能力和独立活动能力,可以转至康复机构。他的病例凸显了抗GQ1b抗体综合征和MG重叠时诊断的复杂性,也突出了艾加莫德在治疗重叠性自身免疫性神经肌肉综合征难治性病例方面的治疗潜力。鉴于艾加莫德在该病例中对改善呼吸和运动功能具有快速疗效,显然艾加莫德在复杂的神经肌肉自身免疫性疾病中可以是一种有价值的治疗选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/854c/12185387/8b0862d45266/fimmu-16-1605985-g001.jpg

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