Hospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo, Instituto da Criança, São Paulo, Brazil.
Vitalant Research Institute, Epidemiology, San Francisco, CA, USA; University of California San Francisco Benioff Children's Hospital Oakland, CA, USA.
Biol Blood Marrow Transplant. 2019 Oct;25(10):2103-2109. doi: 10.1016/j.bbmt.2019.06.013. Epub 2019 Jun 20.
Sickle cell disease (SCD) is associated with significant morbidity, and allogeneic hematopoietic stem cell transplantation (HSCT) remains the primary curative treatment. Recently, the Brazilian Ministry of Health released a regulation that required the publically funded healthcare system to pay for HSCT for SCD patients with defined indications. We used an existing 2794-member SCD cohort established during 2013 to 2015 to characterize candidates for HSCT and estimate the number of possible donors. Of 2064 patients with SC anemia (SCA), 152 of 974 children (16%) and 279 of 1090 adults (26%) had at least 1 HSCT indication. The most common indication for transplant was stroke (n = 239) followed by avascular necrosis (n = 96), priapism (n = 82), cerebrovascular disease (n = 55), >2 vaso-occlusive episodes (n = 38), alloantibodies and chronic transfusion therapy (n = 18), and >2 acute chest syndrome episodes (n = 11). Increasing age, number of transfusions, abnormal transcranial Doppler, retinopathy, dactylitis, and use of hydroxyurea were more frequent in the 152 children with an indication for HSCT compared with 822 without (P < .001). Of 152 children and 279 adults meeting the eligibility definition, 77 (50%) and 204 (73%), respectively, had at least 1 non-SCD full sibling who could potentially serve as a donor. In conclusion, in a large cohort of SCA patients, 16% of children and 26% of adults had at least 1 indication for HSCT; these indications were associated with the severity of the disease. This study provides clinical data necessary for estimating the costs and infrastructure that would be required to implement HSCT in a public healthcare system.
镰状细胞病(SCD)与显著的发病率相关,异基因造血干细胞移植(HSCT)仍然是主要的治愈性治疗方法。最近,巴西卫生部发布了一项规定,要求公共资助的医疗保健系统为符合特定条件的 SCD 患者支付 HSCT 费用。我们使用了一个现有的 2794 名 SCD 患者队列,该队列是在 2013 年至 2015 年期间建立的,用于描述 HSCT 的候选者,并估计可能的供者数量。在 2064 名 SCA 患者中,974 名儿童中有 152 名(16%)和 1090 名成年人中有 279 名(26%)至少有 1 项 HSCT 适应证。移植最常见的适应证是中风(n=239),其次是缺血性坏死(n=96)、阴茎异常勃起(n=82)、脑血管疾病(n=55)、>2 次血管阻塞发作(n=38)、同种异体抗体和慢性输血治疗(n=18)以及>2 次急性胸痛综合征发作(n=11)。在有 HSCT 适应证的 152 名儿童中,与没有适应证的 822 名儿童相比,年龄较大、输血次数较多、异常经颅多普勒、视网膜病变、指骨炎和使用羟基脲的情况更为常见(P<.001)。在符合入选标准的 152 名儿童和 279 名成年人中,分别有 77 名(50%)和 204 名(73%)至少有 1 名非 SCD 全同胞可作为供者。总之,在一个大型 SCA 患者队列中,16%的儿童和 26%的成人至少有 1 项 HSCT 适应证;这些适应证与疾病的严重程度有关。本研究提供了在公共医疗保健系统中实施 HSCT 所需的成本和基础设施的临床数据。