Mariam A Mostafa , Sheref Elseidy , Reham Ali Metwally , Amir Mahmoud , Ali Abdelhay , Farhan S. Imran
{"title":"First reported use of immune checkpoint inhibitor for treatment of cancer in a patient with acquired hemophilia A","authors":"Mariam A Mostafa , Sheref Elseidy , Reham Ali Metwally , Amir Mahmoud , Ali Abdelhay , Farhan S. Imran","doi":"10.1016/j.clicom.2024.04.001","DOIUrl":null,"url":null,"abstract":"<div><p>Acquired hemophilia A is a rare but serious bleeding disorder that occurs because of neutralizing autoantibodies, also called inhibitors that target coagulation factor VIII (FVIII). Although it is a rare disorder, it has high morbidity and mortality with serious, sometimes life-threatening bleeding, often occurring. Immunotherapy with immune checkpoint inhibitors (ICI) is now a key pillar in treatment of malignancies. They have improved outcomes in malignancy but given their mechanism of action, which stimulates the immune response, autoimmune-associated adverse effects are a concern. Several case reports have identified a risk of AHA occurrence in patients treated with ICI. There are no case reports documenting the use or outcomes of ICI in patients with pre-existing AHA. Here we present the first ever case of a patient with AHA in complete remission treated successfully with ICI for lung cancer without relapse in AHA.</p></div>","PeriodicalId":100269,"journal":{"name":"Clinical Immunology Communications","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2024-04-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2772613424000076/pdfft?md5=7a969c587bdd6b21a30b1d5cc76f5f5e&pid=1-s2.0-S2772613424000076-main.pdf","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Clinical Immunology Communications","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2772613424000076","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
Abstract
Acquired hemophilia A is a rare but serious bleeding disorder that occurs because of neutralizing autoantibodies, also called inhibitors that target coagulation factor VIII (FVIII). Although it is a rare disorder, it has high morbidity and mortality with serious, sometimes life-threatening bleeding, often occurring. Immunotherapy with immune checkpoint inhibitors (ICI) is now a key pillar in treatment of malignancies. They have improved outcomes in malignancy but given their mechanism of action, which stimulates the immune response, autoimmune-associated adverse effects are a concern. Several case reports have identified a risk of AHA occurrence in patients treated with ICI. There are no case reports documenting the use or outcomes of ICI in patients with pre-existing AHA. Here we present the first ever case of a patient with AHA in complete remission treated successfully with ICI for lung cancer without relapse in AHA.
获得性血友病 A 是一种罕见但严重的出血性疾病,是由于针对凝血因子 VIII(FVIII)的中和自身抗体(也称为抑制剂)引起的。虽然这是一种罕见的疾病,但它的发病率和死亡率很高,经常会发生严重出血,有时甚至危及生命。免疫检查点抑制剂(ICI)免疫疗法目前已成为治疗恶性肿瘤的关键支柱。它们改善了恶性肿瘤的治疗效果,但由于其作用机制是刺激免疫反应,因此与自身免疫相关的不良反应令人担忧。一些病例报告发现,接受 ICI 治疗的患者有发生 AHA 的风险。目前还没有病例报告记录 ICI 在已有 AHA 的患者中的使用情况或结果。在此,我们介绍了有史以来第一例完全缓解的 AHA 患者在接受 ICI 治疗肺癌后未复发 AHA 的成功病例。