Park Sooyong, Kim Hyun Kyung
Department of Laboratory Medicine, Chungnam National University Hospital, Daejeon, Republic of Korea.
Department of Laboratory Medicine and Cancer Research Institute, Seoul National University College of Medicine, Seoul, Republic of Korea.
Ann Hematol. 2025 May 22. doi: 10.1007/s00277-025-06406-x.
Pure red cell aplasia (PRCA) is a rare hematologic syndrome characterized by anemia with marked reticulocytopenia and, in Asia, is often accompanied by T-cell large granular lymphocyte leukemia (T-LGL). Minimal research has been done on the epidemiology and sequential events of PRCA combined with T-LGL. This study identified 2801 PRCA and 840 T-LGL patients by using big data of the National Health Insurance Service between 2003 and 2022. The average annual crude incidence of PRCA was 2.77 per million and remained stable over 20 years, while T-LGL incidence was 0.82 per million with an increasing trend, possibly reflecting improved diagnostic accessibility. The average age for PRCA and T-LGL onset increased over the study period, consistent with aged society. Associated PRCA conditions are rheumatic diseases (10.5%), thymoma (4.7%), parvovirus infection (1.0%), inflammatory bowel diseases (0.8%), T-LGL (0.6%) and no specific cause (82.4%). Among 18 patients with both PRCA and T-LGL, PRCA preceded T-LGL (50%) or diagnosed concurrently (44%), suggesting that autoreactive T cells in PRCA which suppress erythropoiesis and sequentially evolve into clonal T cell proliferation and, eventually, T-LGL occurrence. This observation supports the hypothesis that both conditions might share a common pathogenic pathway. Further study should identify the causal relationship of PRCA diagnosis followed by T-LGL diagnosis.
纯红细胞再生障碍性贫血(PRCA)是一种罕见的血液学综合征,其特征为贫血伴显著的网织红细胞减少,在亚洲,常伴有T细胞大颗粒淋巴细胞白血病(T-LGL)。关于PRCA合并T-LGL的流行病学和相继发生事件的研究极少。本研究利用2003年至2022年期间国家健康保险服务的大数据确定了2801例PRCA患者和840例T-LGL患者。PRCA的年平均粗发病率为每百万人2.77例,在20年期间保持稳定,而T-LGL的发病率为每百万人0.82例,呈上升趋势,这可能反映了诊断可及性的提高。在研究期间,PRCA和T-LGL发病的平均年龄有所增加,这与老龄化社会相符。与PRCA相关的疾病有风湿性疾病(10.5%)、胸腺瘤(4.7%)、细小病毒感染(1.0%)、炎症性肠病(0.8%)、T-LGL(0.6%)以及无特定病因(82.4%)。在18例同时患有PRCA和T-LGL的患者中,PRCA先于T-LGL发病(50%)或同时诊断(44%),这表明PRCA中抑制红细胞生成的自身反应性T细胞会依次演变为克隆性T细胞增殖,并最终导致T-LGL的发生。这一观察结果支持了这两种疾病可能共享一条共同致病途径的假说。进一步的研究应确定PRCA诊断后再发生T-LGL诊断的因果关系。