Srinivasan M, Taylor A M, Long G V, Herkes G, King G G
Royal North Shore Hospital, Department of Respiratory Medicine, Royal North Shore Hospital, Reserve Rd, St Leonards, 2065, Australia.
Faculty of Medicine and Health, The University of Sydney, Australia.
Respir Med Case Rep. 2021 Oct 21;34:101533. doi: 10.1016/j.rmcr.2021.101533. eCollection 2021.
We present two cases of acute hypercapnic respiratory failure due to diaphragmatic dysfunction secondary to bilateral phrenic nerve paralysis, in patients who were receiving immunotherapy for melanoma. Bilateral diaphragmatic paralysis is an uncommon cause of acute or sub-acute hypercapnic respiratory failure which causes severe breathlessness, orthopnoea and potentially death. Immune checkpoint inhibitors are now standard of care in several solid organ malignancies. However, their use is associated with a risk of developing autoimmune toxicities, which includes mononeuritis. Our two cases demonstrate the potential difficulties in recognising acute hypercapnic respiratory failure and diagnosis of the rare disorder of bilateral diaphragmatic dysfunction, with consequent delays in appropriate management. The occurrence of this rare condition in association with checkpoint inhibitor immunotherapy suggests a possible autoimmune mechanism. Awareness that this rare cause of respiratory failure may occur in patients receiving checkpoint inhibitor therapy might facilitate earlier diagnosis and treatment.
我们报告了两例因双侧膈神经麻痹继发膈肌功能障碍导致急性高碳酸血症性呼吸衰竭的病例，这两名患者正在接受黑色素瘤免疫治疗。双侧膈肌麻痹是急性或亚急性高碳酸血症性呼吸衰竭的罕见原因，可导致严重呼吸困难、端坐呼吸并可能危及生命。免疫检查点抑制剂目前是几种实体器官恶性肿瘤的标准治疗方法。然而，其使用与发生自身免疫毒性的风险相关，其中包括单神经炎。我们的两个病例表明，识别急性高碳酸血症性呼吸衰竭以及诊断罕见的双侧膈肌功能障碍疾病存在潜在困难，从而导致适当治疗的延迟。这种罕见病症与检查点抑制剂免疫治疗相关的情况提示了一种可能的自身免疫机制。认识到接受检查点抑制剂治疗的患者可能出现这种罕见的呼吸衰竭原因，可能有助于早期诊断和治疗。
