Department of Microbiology and Immunology, Miller School of Medicine, University of Miami, Miami, FL, USA.
Immunol Res. 2013 Dec;57(1-3):34-43. doi: 10.1007/s12026-013-8449-0.
Aplastic anemia (AA) is an immune-mediated and life-threatening form of acquired bone marrow failure (BMF), characterized by development and expansion of self-reactive T cells. These T cells cause continuous destruction of hematopoietic stem cells (HSCs), progenitors, and mature blood cells, leading to severe and if left untreated fatal marrow hypoplasia and pancytopenia. Standard treatment options for patients with AA include: (1) immunosuppressive therapy (IST) with anti-thymocyte globulin and cyclosporine A which targets self-reactive T cells, or (2) matched sibling or unrelated BM transplant (BMT). The IST treatment is often not effective due to poor response to therapy or disease relapse after IST. Also, BMT is not an option for many patients due to their age, comorbidities, and the lack of histocompatible donor. This necessitates development and testing of novel approaches to reduce severity of AA and to efficiently treat patients with refractory and relapsed AA. Immune-mediated AA was reproduced in animals, including mouse lymphocyte infusion models, which are used to study further etiology and pathophysiology of AA and test new drugs and approaches in treating and managing AA. In these mouse models the immune correlates and pathologic features of AA are strikingly similar to features of severe human AA. In this article we (a) briefly review standard and developing approaches for treating AA and (b) describe development and testing of novel treatment approach with a potential to safely reduce BM hypoplasia and significantly decrease the loss of HSCs in mouse lymphocyte infusion model of AA.
再生障碍性贫血(AA)是一种免疫介导的、危及生命的获得性骨髓衰竭(BMF)形式,其特征是自身反应性 T 细胞的发展和扩增。这些 T 细胞导致造血干细胞(HSCs)、祖细胞和成熟血细胞的持续破坏,导致严重的、如果不治疗则致命的骨髓发育不良和全血细胞减少症。AA 患者的标准治疗选择包括:(1)针对自身反应性 T 细胞的免疫抑制疗法(IST),包括抗胸腺细胞球蛋白和环孢素 A,或(2)匹配的同胞或无关供体骨髓移植(BMT)。IST 治疗通常效果不佳,因为对治疗反应不佳或 IST 后疾病复发。此外,由于年龄、合并症和缺乏组织相容性供体,许多患者不适合 BMT。这就需要开发和测试新的方法来减轻 AA 的严重程度,并有效地治疗难治性和复发性 AA 患者。免疫介导的 AA 已在动物中复制,包括小鼠淋巴细胞输注模型,用于进一步研究 AA 的病因和病理生理学,并测试治疗和管理 AA 的新药和方法。在这些小鼠模型中,AA 的免疫相关性和病理特征与严重人类 AA 的特征非常相似。在本文中,我们(a)简要回顾了治疗 AA 的标准和发展方法,(b)描述了一种新的治疗方法的开发和测试,该方法具有安全减轻 BM 发育不良和显著减少 AA 小鼠淋巴细胞输注模型中 HSCs 损失的潜力。