Vallée Tanja, Schmid Irene, Gloning Lisa, Bacova Martina, Ahrens Jutta, Feuchtinger Tobias, Klein Christoph, Gaertner Vincent D, Albert Michael H
Department of Pediatrics, Dr. Von Hauner Children's Hospital, University Hospital, LMU Munich, Lindwurmstr. 4, 80337, Munich, Germany.
Ann Hematol. 2023 Nov;102(11):3217-3227. doi: 10.1007/s00277-023-05447-4. Epub 2023 Sep 19.
Many sickle cell disease (SCD) patients lack matched family donors (MFD) or matched unrelated donors (MUD), implying haploidentical donors (MMFD) as a logical donor choice. We used a reduced toxicity protocol for all donor types. We included 31 patients (2-22 years) with MFD (n = 15), MMFD (10), or MUD (6) HSCT and conditioning with alemtuzumab/ATG, thiotepa, fludarabine and treosulfan, and post-transplant cyclophosphamide for MMFD. After the initial six patients, treosulfan was replaced by targeted busulfan (AUC 65-75 ng*h/ml). After a median follow-up of 26 months (6-123), all patients are alive and off immunosuppression. Two MMFD patients experienced secondary graft failure with recurrence of SCD, both after treosulfan conditioning. Neither acute GVHD ≥ °III nor moderate/severe chronic GVHD was observed. The disease-free, severe GVHD-free survival was 100%, 100%, and 80% in the MFD, MUD, and MMFD groups, respectively (p = 0.106). There was a higher rate of virus reactivation in MMFD (100%) and MUD (83%) compared to MFD (40%; p = 0.005), but not of viral disease (20% vs 33% vs 13%; p = 0.576). Six patients had treosulfan-based conditioning, two of whom experienced graft failure (33%), compared to 0/25 (0%) after busulfan-based conditioning (p = 0.032). Donor chimerism was ≥ 80% in 28/31 patients (90%) at last follow-up. Reduced toxicity myeloablative conditioning resulted in excellent overall survival, negligible GVHD, and low toxicity among all donor groups in pediatric and young adult patients with SCD.
许多镰状细胞病(SCD)患者缺乏匹配的家族供体(MFD)或匹配的非亲属供体(MUD),这意味着单倍体相合供体(MMFD)是一种合理的供体选择。我们对所有供体类型都采用了低毒性方案。我们纳入了31例年龄在2至22岁之间的患者,他们接受了MFD(n = 15)、MMFD(10例)或MUD(6例)造血干细胞移植(HSCT),预处理方案为阿仑单抗/抗胸腺细胞球蛋白(ATG)、噻替派、氟达拉滨和曲奥舒凡,MMFD患者在移植后还接受了环磷酰胺治疗。最初的6例患者之后,曲奥舒凡被靶向白消安(AUC 65 - 75 ng*h/ml)替代。经过中位26个月(6 - 123个月)的随访,所有患者均存活且停用了免疫抑制剂。2例MMFD患者出现继发性移植物功能衰竭,镰状细胞病复发,均发生在曲奥舒凡预处理之后。未观察到III°及以上的急性移植物抗宿主病(GVHD),也未观察到中度/重度慢性GVHD。MFD、MUD和MMFD组的无病、无严重GVHD生存率分别为100%、100%和80%(p = 0.106)。与MFD组(40%)相比,MMFD组(100%)和MUD组(83%)的病毒再激活率更高(p = 0.005),但病毒相关疾病发生率无差异(20% vs 33% vs 13%;p = 0.576)。6例患者接受了基于曲奥舒凡的预处理,其中2例出现移植物功能衰竭(33%),而基于白消安的预处理后移植物功能衰竭发生率为0/25(0%)(p = 0.032)。在最后一次随访时,31例患者中有28例(90%)的供体嵌合率≥80%。低毒性清髓性预处理在小儿和年轻成人SCD患者的所有供体组中均带来了出色的总生存率、可忽略不计的GVHD及低毒性。