Sicre de Fontbrune Flore, Chevillon Florian, Fahd Mony, Desseaux Kristell, Poiré Xavier, Forcade Edouard, Sterin Arthur, Neven Bénédicte, Gandemer Virginie, Thepot Sylvain, Garnier Alice, Lioure Bruno, Marcais Ambroise, Nguyen-Quoc Stephanie, Tavitian Suzanne, Vincent Laure, Donadieu Jean, Resche Riggon Matthieu, Chevret Sylvie, Pasquet Marlene, Peffault de Latour Regis
Hématologie Greffe, Hôpital Saint-Louis, APHP, Paris, France.
AJA, Hôpital Saint Louis, APHP, Paris, France.
Br J Haematol. 2024 Aug 19. doi: 10.1111/bjh.19691.
Modalities and timing of haematopoietic stem cell transplant (HSCT) in patients with GATA2 deficiency are still subject to debate. On June 2022, 67 patients (median age 20.6 years) underwent a first allogeneic HSCT among 21 centres. Indications for HSCT were myelodysplastic syndrome (MDS) ≤5% blasts ± immunodeficiency (66%), MDS >5% blasts (15%), acute myeloid leukaemia (19%). Conditioning regimen was myeloablative in 85% and anti-thymocyte globulins were used in 67%. The cumulative incidence (CInc) of acute graft versus host disease (GvHD) grade II-IV and III-IV at day 100 were 42% and 13%, and CInc of chronic and extensive chronic GvHD at 2 years were 42% and 23%. CInc of relapses was 3% and 11% at 1 and 5 years. Overall survival (OS) at 1 and 5 years was 83% and 72% (median follow-up 5.6 years). The factors associated with worse OS in multivariable analysis were the year of HSCT, a history of excess blasts before transplant and peripheral blood stem cell (PBSC) grafts. Age at HSCT, non-myeloablative conditioning and PBSC grafts were associated with increased non-relapse mortality. In conclusion, bone marrow monitoring to identify clonal evolution and perform HSCT before the appearance of excess blast is mandatory.
GATA2 缺乏症患者造血干细胞移植(HSCT)的方式和时机仍存在争议。2022年6月,67例患者(中位年龄20.6岁)在21个中心接受了首次异基因HSCT。HSCT的适应证为骨髓增生异常综合征(MDS)原始细胞≤5% ± 免疫缺陷(66%)、MDS原始细胞>5%(15%)、急性髓系白血病(19%)。85%的患者采用清髓性预处理方案,67%的患者使用了抗胸腺细胞球蛋白。100天时急性移植物抗宿主病(GvHD)Ⅱ - Ⅳ级和Ⅲ - Ⅳ级的累积发生率分别为42%和13%,2年时慢性和广泛性慢性GvHD的累积发生率分别为42%和23%。1年和5年时复发的累积发生率分别为3%和11%。1年和5年时的总生存率(OS)分别为83%和72%(中位随访5.6年)。多变量分析中与较差OS相关的因素为HSCT的年份、移植前原始细胞过多史和外周血干细胞(PBSC)移植。HSCT时的年龄、非清髓性预处理和PBSC移植与非复发死亡率增加相关。总之,进行骨髓监测以识别克隆演变并在原始细胞过多出现之前进行HSCT是必不可少的。