Song Jie, Huan Xiao, Chen Yuanyi, Luo Yeting, Zhong Huahua, Wang Yuan, Yang Lei, Xi Caihua, Yang Yu, Xi Jianying, Zheng Jianming, Wu Zongtai, Zhao Chongbo, Luo Sushan
Huashan Rare Disease Center and Department of Neurology, Huashan Hospital, Shanghai Medical College, National Center for Neurological Disorders, Fudan University, Shanghai, China.
Department of Neurology, Ganzhou People's Hospital, Nanchang University, Nanchang, Jiangxi, China.
Ther Adv Neurol Disord. 2024 Jul 26;17:17562864241261602. doi: 10.1177/17562864241261602. eCollection 2024.
Eculizumab has improved recovery from ventilatory support in myasthenic crisis (MC) cases. However, the safety and efficacy profiles from prospective studies are still lacking. This study aimed to explore eculizumab's safety and efficacy in a prospective case series of patients with refractory MC. We followed a series of anti-acetylcholine receptor (AChR) antibody-positive myasthenia gravis (MG) patients who received eculizumab as an add-on therapy for 12 weeks during MC to facilitate the weaning process and reduced disease activity. Serum anti-AChR antibodies and peripheral immune molecules associated with the complement pathway were evaluated before and after eculizumab administration. Compared to the baseline Myasthenia Gravis Foundation of America (MGFA)-quantitative MG test (QMG) scores (22.25 ± 4.92) and MG-activities of daily living (MG-ADL; 18.25 ± 2.5) scores at crisis, improvements were observed from 4 weeks (14.5 ± 10.47 and 7.5 ± 7.59, respectively) through 12 weeks (7.5 ± 5.74 and 2.25 ± 3.86, respectively) post-treatment. Muscle strength consistently improved across ocular, bulbar, respiratory, and limb/gross domain groups. One patient died of cardiac failure at 16 weeks. Three cases remained in remission at 24 weeks, with a mean QMG score of 2.67 ± 2.89 and ADL score of 0.33 ± 0.58. No significant side effects were reported. Serum CH50 and soluble C5b-9 levels significantly declined, while there were no significant changes in serum anti-AChR antibody levels, C1q, C5a levels, or peripheral lymphocyte proportions. Eculizumab was well tolerated and showed efficacy in this case series. Large prospective cohort studies with extended follow-up periods are needed to further explore the safety and efficacy profile in real-world practice.
依库珠单抗已改善了重症肌无力危象(MC)患者撤机后的恢复情况。然而,前瞻性研究的安全性和疗效数据仍然缺乏。本研究旨在探讨依库珠单抗在难治性MC患者前瞻性病例系列中的安全性和疗效。我们对一系列抗乙酰胆碱受体(AChR)抗体阳性的重症肌无力(MG)患者进行了随访,这些患者在MC期间接受依库珠单抗作为附加治疗12周,以促进撤机过程并降低疾病活动度。在依库珠单抗给药前后评估血清抗AChR抗体和与补体途径相关的外周免疫分子。与基线时美国重症肌无力基金会(MGFA)定量MG测试(QMG)评分(22.25±4.92)和危象时MG日常生活活动(MG-ADL)评分(18.25±2.5)相比,治疗后4周(分别为14.5±10.47和7.5±7.59)至12周(分别为7.5±5.74和2.25±3.86)观察到改善。眼肌、延髓肌、呼吸肌和肢体/全身肌群的肌力持续改善。1例患者在16周时死于心力衰竭。3例患者在24周时仍处于缓解状态,平均QMG评分为2.67±2.89,ADL评分为0.33±0.58。未报告明显副作用。血清CH50和可溶性C5b-9水平显著下降,而血清抗AChR抗体水平、C1q、C5a水平或外周淋巴细胞比例无显著变化。依库珠单抗在本病例系列中耐受性良好且显示出疗效。需要进行更大规模的前瞻性队列研究并延长随访期,以进一步探索其在实际临床中的安全性和疗效。