Department of Paediatrics and Developmental Biology, Tokyo Medical and Dental University Graduate School of Medical and Dental Sciences, Tokyo, Japan.
Br J Haematol. 2011 Aug;154(3):363-72. doi: 10.1111/j.1365-2141.2011.08735.x. Epub 2011 May 14.
We report the results of umbilical cord blood transplantation (UCBT) performed in 88 patients with primary immunodeficiency (PID) between 1998 and 2008 in Japan; severe combined immunodeficiency (SCID, n = 40), Wiskott-Aldrich syndrome (WAS, n = 23), chronic granulomatous disease (n = 7), severe congenital neutropaenia (SCN, n = 5) and other immunodeficiencies (n = 13). Five-year overall survival (5-year OS) for all patients was 69% [95% confidence interval (CI), 57-78%], and was 71% and 82% for SCID and WAS, respectively. The main cause of death before day 100 was infection (17/19), while that after day 100 was graft-versus-host disease (GVHD) (5/7). Using multivariate analyses, pre-transplant infection, no conditioning, ≥ 2 human leucocyte antigen (HLA) mismatches or diagnosis other than SCID, SCN or WAS were all associated with poor prognosis. Reduced-intensity conditioning was associated with decreased overall mortality compared with myeloablative therapy. The cumulative incidence of grade 2-4 acute GVHD at day 100 was 28% (95% CI, 19-38%), and that of chronic GVHD at day 180 was 13% (95% CI, 7-23%). We conclude that UCBT should be considered for PID patients without an HLA-matched sibling. The control of pre-transplant infection and selection of HLA-matched donors will lead to a better outcome.
我们报告了 1998 年至 2008 年间在日本进行的 88 例原发性免疫缺陷(PID)患者脐带血移植(UCBT)的结果;严重联合免疫缺陷(SCID,n=40)、Wiskott-Aldrich 综合征(WAS,n=23)、慢性肉芽肿病(n=7)、严重先天性中性粒细胞减少症(SCN,n=5)和其他免疫缺陷(n=13)。所有患者的 5 年总生存率(5 年 OS)为 69%[95%置信区间(CI),57-78%],SCID 和 WAS 分别为 71%和 82%。100 天前死亡的主要原因是感染(19/19),100 天后死亡的原因是移植物抗宿主病(GVHD)(5/7)。使用多变量分析,移植前感染、无预处理、≥2 个人类白细胞抗原(HLA)错配或诊断为 SCID、SCN 或 WAS 以外的疾病均与预后不良相关。与清髓性治疗相比,减强度预处理与总死亡率降低相关。100 天时 2-4 级急性 GVHD 的累积发生率为 28%(95%CI,19-38%),180 天时慢性 GVHD 的累积发生率为 13%(95%CI,7-23%)。我们得出结论,对于没有 HLA 匹配同胞的 PID 患者,应考虑 UCBT。移植前感染的控制和 HLA 匹配供体的选择将导致更好的结果。