The University of Utah Transplant and Cellular Therapy Program, Salt Lake City, Utah.
Division of Biostatistics, Institute for Heath and Equity, Medical College of Wisconsin, Milwaukee, Wisconsin; Center for International Blood and Marrow Transplant Research), Department of Medicine, Medical College of Wisconsin, Milwaukee, Wisconsin.
Transplant Cell Ther. 2022 Oct;28(10):712.e1-712.e8. doi: 10.1016/j.jtct.2022.07.014. Epub 2022 Jul 18.
Chronic graft-versus-host disease (cGVHD) occurs in up to 25% of children following allogeneic hematopoietic cell transplantation (HCT) and continues to be a major cause of late morbidity and poor quality of life among long-term survivors of pediatric HCT. Late effects (LEs) of HCT are well documented in this population, and cGVHD has been identified as a risk factor for subsequent neoplasms (SNs) and several nonmalignant LEs (NM-LEs); however, the reported correlation between cGVHD and LEs varies among studies. We compared LEs occurring ≥2 years following childhood HCT for a hematologic malignancy in 2-year disease-free survivors with and without cGVHD and further evaluated the association of cGVHD features on the development of LEs. This systematic retrospective analysis used data from the Center of International Blood and Marrow Transplant Research (CIBMTR) on a large, representative cohort of 1260 survivors of pediatric HCT for hematologic malignancy to compare first malignant LEs and NM-LEs in those with a diagnosis of cGVHD and those who never developed cGVHD. The cumulative incidences of any first LE, SN, and NM-LE were estimated at 10 years after HCT, with death as a competing risk for patients with cGVHD versus no cGVHD. Cox proportional hazards models were used to evaluate the impact of cGVHD and its related characteristics on the development of first LEs. The estimated 10-year cumulative incidence of any LE in patients with and without cGVHD was 43% (95% CI, 38% to 48.2%) versus 32% (95% confidence interval [CI], 28.5% to 36.3%) (P < .001), respectively. The development of cGVHD by 2 years post-HCT was independently associated with any LE (hazard ratio [HR], 1.38; 95% CI, 1.13 to 1.68; P = .001) and NM-LE (HR, 1.37; 95% CI, 1.10 to 1.70; P = .006), but not SN (HR, 1.30; 95% CI, .73 to 2.31; P = .38). cGVHD-related factors linked with the development of an NM-LE included having extensive grade cGVHD (HR, 1.60; 95% CI, 1.23 to 2.08; P = .0005), severe cGVHD (HR, 2.25; 95% CI, 1.60 to 3.17; P < .0001), interrupted onset type (HR, 1.57; 95% CI, 1.21 to 2.05; P = .0008), and both mucocutaneous and visceral organ involvement (HR, 1.59; 95% CI, 1.24 to 2.03; P = .0002). No significant association between cGVHD-specific variables and SN was identified. Finally, the duration of cGVHD treatment of cGVHD with systemic immunosuppression was not significantly associated with SNs or NM-LEs. cGVHD was more closely associated with NM-LEs than with SNs among survivors of pediatric HCT for hematologic malignancy. In this analysis, the development of SNs was strongly associated with the use of myeloablative total body irradiation. cGVHD-related characteristics consistent with a state of greater immune dysregulation were more closely linked to NM-LEs.
慢性移植物抗宿主病 (cGVHD) 发生在接受异基因造血细胞移植 (HCT) 的儿童中,高达 25%,并且仍然是儿科 HCT 长期幸存者中晚期发病率和生活质量差的主要原因。在该人群中,HCT 的晚期效应 (LEs) 有充分的记录,cGVHD 已被确定为随后发生肿瘤 (SNs) 和几种非恶性 LEs (NM-LEs) 的危险因素;然而,报告的 cGVHD 与 LEs 之间的相关性在研究中有所不同。我们比较了 2 年内无疾病存活的儿童血液恶性肿瘤 HCT 后≥2 年发生的 LEs,比较了有无 cGVHD 的 2 年疾病无 cGVHD 幸存者,并进一步评估了 cGVHD 特征与 LEs 发生的关系。这项系统的回顾性分析使用了来自国际血液和骨髓移植研究中心 (CIBMTR) 的大型代表性血液恶性肿瘤儿科 HCT 幸存者队列的数据,比较了诊断为 cGVHD 和从未发生 cGVHD 的患者的首次恶性 LEs 和 NM-LEs。cGVHD 与无 cGVHD 患者的任何 LE、SN 和 NM-LE 的累积发生率分别在 HCT 后 10 年时估计为 43%(95%CI,38%至 48.2%)和 32%(95%置信区间 [CI],28.5%至 36.3%)(P<.001)。HCT 后 2 年内发生 cGVHD 与任何 LE(风险比 [HR],1.38;95%CI,1.13 至 1.68;P=0.001)和 NM-LE(HR,1.37;95%CI,1.10 至 1.70;P=0.006)的发生独立相关,但与 SN(HR,1.30;95%CI,0.73 至 2.31;P=0.38)无关。与 NM-LE 发生相关的 cGVHD 相关因素包括广泛的 cGVHD 分级(HR,1.60;95%CI,1.23 至 2.08;P=0.0005)、严重的 cGVHD(HR,2.25;95%CI,1.60 至 3.17;P<.0001)、中断发病类型(HR,1.57;95%CI,1.21 至 2.05;P=0.0008)和黏膜和内脏器官受累(HR,1.59;95%CI,1.24 至 2.03;P=0.0002)。未发现 cGVHD 特异性变量与 SN 之间存在显著相关性。最后,用全身免疫抑制治疗 cGVHD 的 cGVHD 持续时间与 SNs 或 NM-LEs 无显著相关性。在接受血液恶性肿瘤 HCT 的儿童幸存者中,cGVHD 与 NM-LEs 的关系比与 SNs 的关系更为密切。在这项分析中,SNs 的发生与使用全身照射的骨髓清除密切相关。与更严重的免疫失调状态相关的 cGVHD 相关特征与 NM-LEs 更为密切相关。