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中重型再生障碍性贫血的鉴别特征与共性。

Distinctive and common features of moderate aplastic anaemia.

机构信息

Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic, Cleveland, OH, USA.

Leukemia Program, Department of Hematology and Medical Oncology, Taussig Cancer Institute, Cleveland Clinic, Cleveland, OH, USA.

出版信息

Br J Haematol. 2020 Jun;189(5):967-975. doi: 10.1111/bjh.16460. Epub 2020 Jan 31.

Abstract

The therapy algorithm for severe aplastic anaemia (sAA) is established but moderate AA (mAA), which likely reflects a more diverse pathogenic mechanism, often represents a treatment/management conundrum. A cohort of AA patients (n = 325) was queried for those with non-severe disease using stringent criteria including bone marrow hypocellularity and chronic persistence of moderately depressed blood counts. As a result, we have identified and analyzed pathological and clinical features in 85 mAA patients. Progression to sAA and direct clonal evolution (paroxysmal nocturnal haemoglobinuria/acute myeloid leukaemia; PNH/AML) occurred in 16%, 11% and 1% of mAA cases respectively. Of the mAA patients who received immunosuppressive therapy, 67% responded irrespective of time of initiation of therapy while conservatively managed patients showed no spontaneous remissions. Genomic analysis of mAA identified evidence of clonal haematopoiesis with both persisting and remitting patterns at low allelic frequencies; with more pronounced mutational burden in sAA. Most of the mAA patients have autoimmune pathogenesis similar to those with sAA, but mAA contains a mix of patients with diverse aetiologies. Although progression rates differed between mAA and sAA (P = 0·003), cumulative incidences of mortalities were only marginally different (P = 0·095). Our results provide guidance for diagnosis/management of mAA, a condition for which no current standard of care is established.

摘要

再生障碍性贫血(AA)的治疗方案已经建立,但中度 AA(mAA)可能反映了更具多样性的发病机制,通常代表了治疗/管理的难题。我们对一组 AA 患者(n=325)进行了查询,以确定那些符合严格标准(包括骨髓细胞减少症和中度血细胞计数持续减少)的非严重疾病患者。结果,我们在 85 例 mAA 患者中确定并分析了病理和临床特征。mAA 患者分别有 16%、11%和 1%进展为 sAA 和直接克隆进化(阵发性睡眠性血红蛋白尿/急性髓系白血病;PNH/AML)。接受免疫抑制治疗的 mAA 患者中,67%的患者有反应,而不管治疗开始的时间如何,而保守治疗的患者则没有自发缓解。mAA 的基因组分析发现了克隆性造血的证据,具有持续和缓解模式,等位基因频率较低;sAA 的突变负担更为明显。大多数 mAA 患者具有与 sAA 相似的自身免疫发病机制,但 mAA 包含了具有不同病因的患者混合。尽管 mAA 和 sAA 之间的进展率不同(P=0·003),但死亡率的累积发生率仅略有差异(P=0·095)。我们的研究结果为 mAA 的诊断/管理提供了指导,目前尚未确定 mAA 的标准治疗方法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1ba0/9290125/24f631bd8c42/BJH-189-967-g002.jpg

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