Algeri Mattia, Comoli Patrizia, Strocchio Luisa, Perotti Cesare, Corbella Franco, Del Fante Claudia, Baio Ambrogia, Giorgiani Giovanna, Gurrado Antonella, Accornero Elisa, Cugno Chiara, Pession Andrea, Zecca Marco
*Oncoematologia Pediatrica ‡Struttura Complessa di Radioterapia Oncologica, Fondazione IRCCS Policlinico San Matteo †Servizio di Immunoematologia e Medicina Trasfusionale, Fondazione IRCCS Policlinico San Matteo, Pavia §Clinica Pediatrica, Policlinico Sant'Orsola, Bologna, Italy.
J Pediatr Hematol Oncol. 2015 May;37(4):322-6. doi: 10.1097/MPH.0000000000000283.
Allogeneic hematopoietic stem cell transplantation (HSCT) is the only cure for marrow failure associated with dyskeratosis congenita (DC). Data on transplants from alternative donors are limited. We describe a boy with DC and severe aplastic anemia who underwent haploidentical T-cell depleted HSCT using a reduced-intensity conditioning regimen. He underwent engraftment without toxicity or GVHD. His posttransplant course was complicated by EBV reactivation, treated with rituximab and EBV-specific T lymphocytes. After 26 months, he is in complete chimerism, with normal blood count and no sign of GVHD or pulmonary dysfunction. To the best of our knowledge, this is the first report of DC successfully treated with allogeneic HSCT from a haploidentical family donor.
异基因造血干细胞移植(HSCT)是治疗先天性角化不良(DC)相关骨髓衰竭的唯一方法。来自替代供体的移植数据有限。我们描述了一名患有DC和严重再生障碍性贫血的男孩,他接受了使用低强度预处理方案的单倍体相合T细胞去除HSCT。他实现了植入,且无毒性或移植物抗宿主病(GVHD)。他的移植后病程因EB病毒重新激活而复杂化,通过利妥昔单抗和EB病毒特异性T淋巴细胞进行治疗。26个月后,他处于完全嵌合状态,血细胞计数正常,没有GVHD或肺功能障碍的迹象。据我们所知,这是首例成功通过单倍体相合家族供体的异基因HSCT治疗DC的报告。