Baptista Carlota, Margarido Inês, Bizarro Rita, Branco Francisco P, Faria Ana
Medical Oncology, Hospital Beatriz Ângelo, Loures, PRT.
Medical Oncology, Hospital da Luz, Lisbon, PRT.
Cureus. 2025 Jan 2;17(1):e76834. doi: 10.7759/cureus.76834. eCollection 2025 Jan.
Triple M Overlap Syndrome (TMOS) is a rare and severe complication of immune checkpoint inhibitor (ICI) therapy, characterized by concurrent myocarditis, myositis and myasthenia gravis. There is a scarcity of evidence regarding this syndrome, underscoring the need for further research and reporting. We report the case of a woman in her 70s with stage IV microsatellite instability-high (MSI-H) colon cancer who developed generalized myalgias and muscle weakness, along with bulbar and ocular symptoms, two weeks after the first administration of pembrolizumab. Laboratory tests revealed elevated creatine kinase, transaminase and troponin levels. Cardiac magnetic resonance imaging (MRI) findings were suggestive of myocarditis. Due to the constellation of myocarditis, myositis and myasthenia gravis, TMOS was diagnosed. Despite initial improvement with corticosteroid pulses, the patient's clinical condition deteriorated, culminating in the need for mechanical ventilation due to respiratory failure. Sequential treatment with intravenous immunoglobulin (IVIG) and, ultimately, plasmapheresis resulted in gradual clinical improvement. After a prolonged Intensive Care Unit (ICU) stay, she was admitted to the medical ward, where she continued to improve with speech therapy and motor rehabilitation. ICI therapy was permanently discontinued. A high level of suspicion is necessary for the diagnosis of this rare syndrome, and multidisciplinary collaboration is critical to optimize clinical outcomes. This case highlights the importance of evaluating myocarditis, myositis and myasthenia when any of them is diagnosed in a patient receiving ICIs. Prompt treatment initiation and ICI therapy discontinuation are crucial to maximizing the chances of a positive outcome.
三肌重叠综合征(TMOS)是免疫检查点抑制剂(ICI)治疗的一种罕见且严重的并发症,其特征为同时发生心肌炎、肌炎和重症肌无力。关于该综合征的证据匮乏,这凸显了进一步研究和报告的必要性。我们报告了一例70多岁患有IV期微卫星高度不稳定(MSI-H)结肠癌的女性病例,该患者在首次使用帕博利珠单抗两周后出现全身肌痛、肌肉无力以及延髓和眼部症状。实验室检查显示肌酸激酶、转氨酶和肌钙蛋白水平升高。心脏磁共振成像(MRI)结果提示心肌炎。由于存在心肌炎、肌炎和重症肌无力的综合表现,该患者被诊断为TMOS。尽管最初使用糖皮质激素冲击治疗后病情有所改善,但患者的临床状况仍恶化,最终因呼吸衰竭需要机械通气。随后依次使用静脉注射免疫球蛋白(IVIG),最终进行血浆置换,患者的临床症状逐渐改善。在重症监护病房(ICU)长时间住院后,她被转入内科病房,在那里通过言语治疗和运动康复继续好转。ICI治疗被永久停用。对于这种罕见综合征的诊断,高度怀疑很有必要,多学科协作对于优化临床结果至关重要。该病例强调了在接受ICI治疗的患者中诊断出任何一种情况时,评估心肌炎、肌炎和重症肌无力的重要性。及时开始治疗并停用ICI治疗对于最大化获得良好结果的机会至关重要。