Hemophagocytic lymphohistiocytosis associated with immune checkpoint inhibitor use: A review of the current knowledge and future directions

IF 2.1 4区 医学 Q3 HEMATOLOGY Blood Cells Molecules and Diseases Pub Date : 2024-09-30 DOI:10.1016/j.bcmd.2024.102896
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Abstract

Hemophagocytic lymphohistiocytosis (HLH) is a severe and often lethal inflammatory syndrome characterized by excessive immune activation leading to fever, cytopenias, and multiorgan involvement. Immune checkpoint inhibitors (ICIs) are central to many contemporary cancer regimens, but their use is associated with immune-related adverse events. Here, we report a case of ICI-induced HLH successfully treated with single agent dexamethasone and provide a scoping review of the literature for cases of ICI-induced HLH with a focus on treatment strategies and outcomes. Using the Medline database, we searched for cases of ICI-associated HLH, with a total of 51 cases reported between 2017 and 2023. Our results underscore the severe nature of this disease, with a 13.7 % mortality rate across 51 case reports. Treatment strategies for ICI-induced HLH were variable: steroids alone (56.9 %), steroids with etoposide (17.6 %), steroids with tociluzumab (11.8 %), among other combinations. Our literature review indicates that steroids alone may be sufficient treatment in some cases of ICI-HLH, with comparable mortality with steroids alone (n = 29) (13.8 %) to that of cases treated with both steroids and immunomodulators (n = 15, 13.3 %). Moreover, all patients treated with steroids and tocilizumab survived (n = 6), suggesting that tocilizumab may be a reasonable next line of therapy when steroid monotherapy proves inadequate. We propose an outline for investigation and treatment of this rare complication of ICI use. Finally, we discuss possible future approaches to develop evidence-based strategies for the diagnosis and management of ICI-induced HLH including the importance of integrating the role of patient community involvement.
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与使用免疫检查点抑制剂相关的嗜血细胞淋巴组织细胞增多症:回顾当前知识和未来方向。
嗜血细胞淋巴组织细胞增多症(HLH)是一种严重的炎症综合征,通常是致命的,其特点是过度免疫激活导致发热、细胞减少和多器官受累。免疫检查点抑制剂(ICIs)是许多当代癌症治疗方案的核心,但其使用与免疫相关不良事件有关。在此,我们报告了一例单药地塞米松成功治疗 ICI 诱导的 HLH 病例,并对 ICI 诱导的 HLH 病例进行了文献综述,重点关注治疗策略和结果。我们使用 Medline 数据库搜索了 ICI 相关 HLH 病例,2017 年至 2023 年间共报道了 51 例。我们的研究结果凸显了这种疾病的严重性,51 例病例报告中的死亡率为 13.7%。ICI诱发的HLH的治疗策略各不相同:单纯类固醇(56.9%)、类固醇联合依托泊苷(17.6%)、类固醇联合托珠单抗(11.8%)以及其他组合。我们的文献综述显示,在某些 ICI-HLH 病例中,单用类固醇治疗可能就足够了,单用类固醇治疗的死亡率(29 例)(13.8%)与同时使用类固醇和免疫调节剂治疗的死亡率(15 例,13.3%)相当。此外,所有接受类固醇和托珠单抗治疗的患者都存活了下来(n = 6),这表明当类固醇单药治疗效果不佳时,托珠单抗可能是合理的下一步治疗方案。我们就 ICI 罕见并发症的调查和治疗提出了建议。最后,我们讨论了未来可能采取的方法,为 ICI 诱导的 HLH 的诊断和管理制定循证策略,包括整合患者社区参与的重要性。
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来源期刊
CiteScore
4.90
自引率
0.00%
发文量
42
审稿时长
14 days
期刊介绍: Blood Cells, Molecules & Diseases emphasizes not only blood cells, but also covers the molecular basis of hematologic disease and studies of the diseases themselves. This is an invaluable resource to all those interested in the study of hematology, cell biology, immunology, and human genetics.
期刊最新文献
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