Kawakami Toru, Kawakami Fumihiro, Matsuzawa Shuji, Yamane Taku, Mizuno Yuga, Asakura Ami, Higano Daigo, Miyairi Shotaro, Sakai Kaoko, Nishina Sayaka, Sakai Hitoshi, Kubota Yasushi, Higuchi Yumiko, Nakazawa Hideyuki, Ishida Fumihiro
Department of Hematology and Clinical Oncology, Shinshu University School of Medicine, Matsumoto, Japan.
Central Laboratory Department, Shinshu University Hospital, Matsumoto, Japan.
Ann Hematol. 2025 Mar;104(3):1471-1479. doi: 10.1007/s00277-025-06356-4. Epub 2025 Apr 9.
Dysregulation of T cell-mediated immunity is considered a major pathophysiological mechanism in acquired pure red cell aplasia (PRCA), including idiopathic PRCA, large granular lymphocytic leukemia-associated PRCA, and thymoma-associated PRCA. Although STAT3 mutations are frequently detected in PRCA patients, the roles of other mutational profiles and their impact on clinical characteristics remain unclear. In this study, whole-exome sequencing and targeted sequencing using a custom-designed panel were performed on 53 PRCA patients. The most frequently mutated genes were STAT3 (36%), PCLO (9%), TET2 (9%), NEB (6%), DNMT3A (6%), and POT1 (6%). Based on genetic profiles, patients were classified into three groups: those with STAT3 variants (group S), those without STAT3 variants but with variants in clonal hematopoiesis (CH)-related genes (group C), and those without variants in either STAT3 or CH-related genes (group O). Patients in group O had a higher median age compared to group S, while group S exhibited milder anemia severity than group C. Additionally, POT1 variants were associated with the idiopathic subtype of PRCA in females, often co-occurring with STAT3 variants. Variants in CH-related genes and other genes, including STAT3 and POT1, may play crucial roles in the pathophysiology of PRCA.
T细胞介导的免疫失调被认为是获得性纯红细胞再生障碍性贫血(PRCA)的主要病理生理机制,包括特发性PRCA、大颗粒淋巴细胞白血病相关PRCA和胸腺瘤相关PRCA。尽管在PRCA患者中经常检测到STAT3突变,但其他突变谱的作用及其对临床特征的影响仍不清楚。在本研究中,对53例PRCA患者进行了全外显子测序和使用定制设计的基因panel进行靶向测序。最常发生突变的基因是STAT3(36%)、PCLO(9%)、TET2(9%)、NEB(6%)、DNMT3A(6%)和POT1(6%)。根据基因谱,患者被分为三组:有STAT3变异的患者(S组)、无STAT3变异但有克隆性造血(CH)相关基因变异的患者(C组),以及无STAT3或CH相关基因变异的患者(O组)。与S组相比,O组患者的年龄中位数更高,而S组的贫血严重程度比C组轻。此外,POT1变异与女性特发性PRCA亚型相关,常与STAT3变异同时出现。CH相关基因和其他基因(包括STAT3和POT1)的变异可能在PRCA的病理生理过程中起关键作用。