Department Blood and Marrow Transplantation, Children's Healthcare of Atlanta, Aflac Cancer and Blood Disorders Center, Atlanta, Georgia.
Spanish Bone Marrow Donor Registry, Josep Carreras Foundation and Leukemia Research Institute, Barcelona, Catalunya, Spain.
Transplant Cell Ther. 2023 Mar;29(3):151-163. doi: 10.1016/j.jtct.2022.11.015. Epub 2022 Nov 25.
Transplantation-associated thrombotic microangiopathy (TA-TMA) is an increasingly recognized complication of hematopoietic cell transplantation (HCT) associated with significant morbidity and mortality. However, TA-TMA is a clinical diagnosis, and multiple criteria have been proposed without universal application. Although some patients have a self-resolving disease, others progress to multiorgan failure and/or death. Poor prognostic features also are not uniformly accepted. The lack of harmonization of diagnostic and prognostic markers has precluded multi-institutional studies to better understand incidence and outcomes. Even current interventional trials use different criteria, making it challenging to interpret the data. To address this urgent need, the American Society for Transplantation and Cellular Therapy, Center for International Bone Marrow Transplant Research, Asia-Pacific Blood and Marrow Transplantation, and European Society for Blood and Marrow Transplantation nominated representatives for an expert panel tasked with reaching consensus on diagnostic and prognostic criteria. The panel reviewed literature, generated consensus statements regarding diagnostic and prognostic features of TA-TMA using the Delphi method, and identified future directions of investigation. Consensus was reached on 4 key concepts: (1) TA-TMA can be diagnosed using clinical and laboratory criteria or tissue biopsy of kidney or gastrointestinal tissue; however, biopsy is not required; (2) consensus diagnostic criteria are proposed using the modified Jodele criteria with additional definitions of anemia and thrombocytopenia. TA-TMA is diagnosed when ≥4 of the following 7 features occur twice within 14 days: anemia, defined as failure to achieve transfusion independence despite neutrophil engraftment; hemoglobin decline by ≥1 g/dL or new-onset transfusion dependence; thrombocytopenia, defined as failure to achieve platelet engraftment, higher-than-expected transfusion needs, refractory to platelet transfusions, or ≥50% reduction in baseline platelet count after full platelet engraftment; lactate dehydrogenase (LDH) exceeding the upper limit of normal (ULN); schistocytes; hypertension; soluble C5b-9 (sC5b-9) exceeding the ULN; and proteinuria (≥1 mg/mg random urine protein-to-creatinine ratio [rUPCR]); (3) patients with any of the following features are at increased risk of nonrelapse mortality and should be stratified as high-risk TA-TMA: elevated sC5b-9, LDH ≥2 times the ULN, rUPCR ≥1 mg/mg, multiorgan dysfunction, concurrent grade II-IV acute graft-versus-host disease (GVHD), or infection (bacterial or viral); and (4) all allogeneic and pediatric autologous HCT recipients with neuroblastoma should be screened weekly for TA-TMA during the first 100 days post-HCT. Patients diagnosed with TA-TMA should be risk-stratified, and those with high-risk disease should be offered participation in a clinical trial for TA-TMA-directed therapy if available. We propose that these criteria and risk stratification features be used in data registries, prospective studies, and clinical practice across international settings. This harmonization will facilitate the investigation of TA-TMA across populations diverse in race, ethnicity, age, disease indications, and transplantation characteristics. As these criteria are widely used, we expect continued refinement as necessary. Efforts to identify more specific diagnostic and prognostic biomarkers are a top priority of the field. Finally, an investigation of the impact of TA-TMA-directed treatment, particularly in the setting of concurrent highly morbid complications, such as steroid-refractory GVHD and infection, is critically needed.
移植相关血栓性微血管病(TA-TMA)是造血细胞移植(HCT)相关的一种日益被认识到的并发症,与较高的发病率和死亡率相关。然而,TA-TMA 是一种临床诊断,提出了多种标准,但并未得到普遍应用。尽管有些患者的疾病可以自行缓解,但有些患者会进展为多器官衰竭和/或死亡。预后不良的特征也未被普遍接受。缺乏对诊断和预后标志物的协调一致,使得无法开展多机构研究来更好地了解发病率和结局。甚至目前的介入性试验也使用不同的标准,使得数据的解释具有挑战性。为了解决这一紧迫需求,美国移植和细胞治疗学会、国际骨髓移植研究中心、亚太血液和骨髓移植学会以及欧洲血液和骨髓移植学会提名代表组成一个专家小组,负责就诊断和预后标准达成共识。该小组审查了文献,使用 Delphi 方法就 TA-TMA 的诊断和预后特征达成了共识声明,并确定了未来的研究方向。专家组就以下 4 个关键概念达成共识:(1)可以使用临床和实验室标准或肾脏或胃肠道组织的组织活检来诊断 TA-TMA;但是,不需要进行活检。(2)使用改良的 Jodele 标准并附加贫血和血小板减少症的定义提出了共识诊断标准。当以下 7 个特征中的 4 个在 14 天内出现两次时,可以诊断 TA-TMA:贫血,定义为尽管中性粒细胞植入但仍无法实现输血独立性;血红蛋白下降≥1 g/dL 或新出现输血依赖;血小板减少症,定义为未达到血小板植入、高于预期的输血需求、对血小板输注无反应或在完全血小板植入后基线血小板计数下降≥50%;乳酸脱氢酶(LDH)超过正常值上限(ULN);裂体细胞;高血压;可溶性 C5b-9(sC5b-9)超过 ULN;蛋白尿(随机尿蛋白与肌酐比值≥1 mg/mg [rUPCR])。(3)具有以下任何特征的患者发生非复发死亡率的风险增加,应被归类为高危 TA-TMA:sC5b-9 升高、LDH 超过 ULN 的 2 倍、rUPCR≥1 mg/mg、多器官功能障碍、同时发生 2 级或 4 级急性移植物抗宿主病(GVHD)或感染(细菌或病毒)。(4)所有接受同种异体和儿科自体 HCT 的神经母细胞瘤患者,在 HCT 后 100 天内应每周筛查 TA-TMA。诊断为 TA-TMA 的患者应进行风险分层,如果有可用的 TA-TMA 靶向治疗临床试验,应向高危疾病患者提供参与该试验的机会。我们建议在国际环境中,使用这些标准和风险分层特征在数据登记处、前瞻性研究和临床实践中进行。这种协调将促进对不同种族、民族、年龄、疾病指征和移植特征的人群中的 TA-TMA 进行研究。随着这些标准的广泛应用,我们预计会根据需要进行持续改进。确定更特异的诊断和预后生物标志物是该领域的首要任务。最后,迫切需要研究 TA-TMA 靶向治疗的影响,特别是在同时存在高发病率并发症的情况下,如类固醇难治性 GVHD 和感染。