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肾细胞癌患者中与双重免疫检查点抑制剂相关的膈肌肌病

Diaphragmatic Myopathy Associated with Dual Immune Checkpoint Inhibitors in a Patient with Renal Cell Carcinoma.

作者信息

Walder Jeremy R, Faiz Saadia A, Tummala Sudhakar, Weathers Shiao-Pei, Palaskas Nicolas L, Buni Maryam, Sheshadri Ajay, Bashoura Lara

机构信息

Divisions of Critical Care, Pulmonary and Sleep Medicine, McGovern Medical School at UTHealth, Houston, TX, USA.

Department of Pulmonary Medicine, The University of Texas MD Anderson Cancer Center, Houston, TX, USA.

出版信息

J Immunother Precis Oncol. 2024 Nov 17;7(4):304-307. doi: 10.36401/JIPO-23-45. eCollection 2024 Nov.

DOI:10.36401/JIPO-23-45
PMID:39553443
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11541929/
Abstract

Immune-related adverse events (irAEs) have become increasingly prevalent with immune checkpoint inhibitor (ICI) cancer treatment. We present a 79-year-old man with metastatic renal cell carcinoma who developed shortness of breath and hypercapnic respiratory insufficiency after his first cycle of nivolumab and ipilimumab. Laboratory data showed elevated creatinine kinase, troponins, and transaminases. Computed tomography of the chest demonstrated bilateral lower lobe atelectasis. Heart catheterization and endomyocardial biopsy were unremarkable. Electromyogram (EMG) and nerve conduction studies (NCS) of the limb muscles revealed mild diffuse myopathy, normal sensory nerve conductions, and low-amplitude motor responses. Subsequent diaphragmatic EMG and NCS demonstrated severe myopathy. ICI-mediated myopathy predominantly affecting diaphragmatic muscles was diagnosed. Treatment included intravenous methylprednisolone, infliximab, abatacept, rituximab, and plasmapheresis. He underwent tracheostomy placement on hospital day 11 due to minimal improvement. He was discharged to a long-term acute care hospital, but unfortunately, he died less than 1 month later due to recurrent infections. irAEs can affect any organ system, but diaphragmatic dysfunction is uncommon. Use of diaphragmatic EMG, NCS, ultrasound study, or biopsy can support the diagnosis. Treatment includes systemic steroids, plasmapheresis, immunosuppressive medications, respiratory support, and cessation of causative medications. ICI-related diaphragmatic dysfunction should be suspected in those patients at risk with hypoxia, hypercapnia, or prolonged invasive or noninvasive ventilation without a distinct etiology. This case report exemplifies the importance of multidisciplinary workup and management of respiratory symptoms and insufficiency to identify and ameliorate irAEs. Diaphragmatic involvement can be associated with significant morbidity and mortality despite early aggressive multimodal therapy.

摘要

免疫相关不良事件(irAEs)在免疫检查点抑制剂(ICI)癌症治疗中变得越来越普遍。我们报告一名79岁的转移性肾细胞癌男性患者,在接受第一个周期的纳武单抗和伊匹单抗治疗后出现呼吸急促和高碳酸血症性呼吸功能不全。实验室数据显示肌酸激酶、肌钙蛋白和转氨酶升高。胸部计算机断层扫描显示双侧下叶肺不张。心脏导管检查和心内膜活检无异常。肢体肌肉的肌电图(EMG)和神经传导研究(NCS)显示轻度弥漫性肌病、感觉神经传导正常和运动反应幅度低。随后的膈肌EMG和NCS显示严重肌病。诊断为ICI介导的主要影响膈肌的肌病。治疗包括静脉注射甲泼尼龙、英夫利昔单抗、阿巴西普、利妥昔单抗和血浆置换。由于改善甚微,他在住院第11天接受了气管造口术。他被转至长期急性护理医院,但不幸的是,他在不到1个月后因反复感染死亡。irAEs可影响任何器官系统,但膈肌功能障碍并不常见。使用膈肌EMG、NCS、超声检查或活检可支持诊断。治疗包括全身用类固醇、血浆置换、免疫抑制药物、呼吸支持和停用致病药物。对于有缺氧、高碳酸血症风险或长期有创或无创通气且无明确病因的患者,应怀疑与ICI相关的膈肌功能障碍。本病例报告例证了多学科检查和处理呼吸症状及功能不全以识别和改善irAEs的重要性。尽管早期积极采用多模式治疗,但膈肌受累仍可能与显著的发病率和死亡率相关。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/31a2350a797c/i2590-017X-7-4-304-f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/acfa7fae05e1/i2590-017X-7-4-304-f01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/0102b378adb0/i2590-017X-7-4-304-f02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/31a2350a797c/i2590-017X-7-4-304-f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/acfa7fae05e1/i2590-017X-7-4-304-f01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/0102b378adb0/i2590-017X-7-4-304-f02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9c5b/11541929/31a2350a797c/i2590-017X-7-4-304-f03.jpg

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本文引用的文献

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Electrodiagnostic and ultrasound evaluation of respiratory weakness.呼吸肌无力的电诊断和超声评估。
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