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免疫相关的心肌炎、肌炎和重症肌无力(MMM)重叠综合征的管理:一项单机构病例系列研究及文献综述

Management of immune-related myocarditis, myositis and myasthenia gravis (MMM) overlap syndrome: a single institution case series and literature review.

作者信息

Sánchez-Camacho Alberto, Torres-Zurita Alberto, Gallego-López Laura, Hernández-Pacheco Rocío, Silva-Romeiro Silvia, Álamo de la Gala María Del Carmen, Peral-Gutiérrez de Ceballos Enrique, de la Cruz-Merino Luis

机构信息

Department of Medical Oncology, Hospital Universitario Virgen Macarena, Seville, Spain.

Autoimmune Disease Unit, Department of Internal Medicine, Hospital Universitario Virgen Macarena, Seville, Spain.

出版信息

Front Immunol. 2025 May 8;16:1597259. doi: 10.3389/fimmu.2025.1597259. eCollection 2025.

Abstract

BACKGROUND

Immune checkpoint inhibitors (ICIs) have revolutionized the treatment of various malignancies, particularly melanoma. However, immune-related adverse events (irAEs) pose significant challenges, particularly in cases of severe toxicity syndromes. One such life-threatening irAE is the myocarditis, myositis, and myasthenia gravis (MMM) overlap syndrome, which occurs in less than 1% of patients but has in-hospital mortality rates ranging from 40 to 60%. Due to its rarity and complexity, early recognition and a multidisciplinary approach are critical to improving outcomes.

METHODS

We present a single-institution case series of four patients diagnosed with MMM overlap syndrome following ICI therapy. Clinical presentation, laboratory findings, imaging, and electrophysiological tests were analyzed to confirm the diagnosis. Therapeutic interventions-including corticosteroids, intravenous immunoglobulins (IVIG), plasma exchange (PLEX), tocilizumab, and rituximab- were evaluated in terms of efficacy and clinical outcomes.

RESULTS

The onset of MMM syndrome varied from 2 to 4 weeks after initiating ICI therapy. Patients presented with rapidly progressive symptoms, including ptosis, bulbar dysfunction, respiratory distress, myopathy, and cardiac conduction abnormalities. Immunosuppressive therapy with high-dose corticosteroids was initiated in all cases. Additional immunomodulatory treatments (IVIG, tocilizumab, PLEX, and rituximab) were administered based on clinical deterioration and autoimmune profile. Two patients achieved complete recovery, one remains on maintenance immunosuppression, and one died due to respiratory failure despite aggressive treatment.

CONCLUSION

MMM overlap syndrome is a severe and often fatal irAE associated with ICI therapy. Early identification, aggressive immunosuppressive treatment, and individualized therapeutic strategies are essential to optimize patient outcomes. Further research is needed to refine diagnostic criteria, identify predictive biomarkers, and establish standardized treatment protocols.

摘要

背景

免疫检查点抑制剂(ICI)彻底改变了各种恶性肿瘤的治疗方式,尤其是黑色素瘤。然而,免疫相关不良事件(irAE)带来了重大挑战,特别是在严重毒性综合征的情况下。一种危及生命的irAE是心肌炎、肌炎和重症肌无力(MMM)重叠综合征,其发生率不到1%,但住院死亡率在40%至60%之间。由于其罕见性和复杂性,早期识别和多学科方法对于改善预后至关重要。

方法

我们报告了一个单中心病例系列,包含4例接受ICI治疗后被诊断为MMM重叠综合征的患者。分析了临床表现、实验室检查结果、影像学和电生理检查以确诊。评估了包括皮质类固醇、静脉注射免疫球蛋白(IVIG)、血浆置换(PLEX)、托珠单抗和利妥昔单抗在内的治疗干预措施的疗效和临床结果。

结果

MMM综合征的发病时间在开始ICI治疗后2至4周不等。患者表现出快速进展的症状,包括上睑下垂、延髓功能障碍、呼吸窘迫、肌病和心脏传导异常。所有病例均开始使用大剂量皮质类固醇进行免疫抑制治疗。根据临床恶化情况和自身免疫特征给予额外的免疫调节治疗(IVIG、托珠单抗、PLEX和利妥昔单抗)。2例患者完全康复,1例仍在接受维持性免疫抑制治疗,1例尽管积极治疗仍因呼吸衰竭死亡。

结论

MMM重叠综合征是一种与ICI治疗相关的严重且通常致命的irAE。早期识别、积极的免疫抑制治疗和个体化的治疗策略对于优化患者预后至关重要。需要进一步研究以完善诊断标准、确定预测生物标志物并建立标准化治疗方案。

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