依库珠单抗治疗肌无力危象的安全性和疗效:前瞻性小型病例系列研究。

IF 4.7 2区 医学 Q1 CLINICAL NEUROLOGY Therapeutic Advances in Neurological Disorders Pub Date : 2024-07-26 eCollection Date: 2024-01-01 DOI:10.1177/17562864241261602
Jie Song, Xiao Huan, Yuanyi Chen, Yeting Luo, Huahua Zhong, Yuan Wang, Lei Yang, Caihua Xi, Yu Yang, Jianying Xi, Jianming Zheng, Zongtai Wu, Chongbo Zhao, Sushan Luo
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引用次数: 0

摘要

依库珠单抗改善了肌无力危象(MC)病例从呼吸机支持中恢复的情况。然而,目前仍缺乏前瞻性研究对其安全性和疗效进行评估。本研究旨在探索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有效性。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期间接受了12周的依库珠单抗作为附加疗法,以促进断奶过程并减少疾病活动。在使用依库珠单抗前后,对血清中的抗ACHR抗体和与补体途径相关的外周免疫分子进行了评估。与危机时的基线美国肌萎缩症基金会(MGFA)-MG定量测试(QMG)评分(22.25 ± 4.92)和MG-日常生活活动(MG-ADL;18.25 ± 2.5)评分相比,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)的评分均有所改善。眼部、球部、呼吸和肢体/总领域组的肌力持续改善。一名患者在 16 周时死于心力衰竭。三例患者在 24 周时病情仍在缓解,平均 QMG 得分为 2.67 ± 2.89,ADL 得分为 0.33 ± 0.58。无明显副作用报告。血清CH50和可溶性C5b-9水平明显下降,而血清抗ACHR抗体水平、C1q、C5a水平或外周淋巴细胞比例均无明显变化。在这一系列病例中,依库珠单抗具有良好的耐受性和疗效。我们需要延长随访期的大型前瞻性队列研究,以进一步探索实际应用中的安全性和有效性。
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The safety and efficacy profile of eculizumab in myasthenic crisis: a prospective small case series.

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.

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来源期刊
CiteScore
8.30
自引率
1.70%
发文量
62
审稿时长
15 weeks
期刊介绍: Therapeutic Advances in Neurological Disorders is a peer-reviewed, open access journal delivering the highest quality articles, reviews, and scholarly comment on pioneering efforts and innovative studies across all areas of neurology. The journal has a strong clinical and pharmacological focus and is aimed at clinicians and researchers in neurology, providing a forum in print and online for publishing the highest quality articles in this area.
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