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儿童、青少年和青年骨髓增生异常肿瘤患者的特征和结局:一项单中心回顾性分析。

Characteristics and outcomes of children, adolescent, and young adult patients with myelodysplastic neoplasms: A single-center retrospective analysis.

机构信息

Department of Pediatrics, The University of Texas MD Anderson Cancer Center, Houston, TX, USA.

Department of Medicine, Baylor University, Houston, TX, USA.

出版信息

Leuk Res. 2024 Sep;144:107563. doi: 10.1016/j.leukres.2024.107563. Epub 2024 Aug 20.

DOI:10.1016/j.leukres.2024.107563
PMID:39178611
Abstract

Myelodysplastic syndrome, or myelodysplastic neoplasms, are a rare finding in pediatric, adolescent, and young adult (AYA) patients. More literature is needed to highlight trends of survival or treatment resistance in subpopulations to improve treatment. Here we report a single center retrospective analysis of pediatric and AYA patients from 2000 to 2022 including molecular and cytogenetic data. Using the IPSS-R and IPSS-M, which have been reported exclusively in adults, and excluding patients with bone marrow failure syndromes, we analyzed 119 pediatric and AYA patients with myelodysplastic neoplasms. Therapy-related myelodysplastic neoplasms were present in 36 % of patients, and 31 % of patients developed acute myeloid leukemia. The 5-year overall survival (OS) rate for the entire cohort was 45 %. Contrary to young adults and older adults, mutations were not common in pediatrics. Those who underwent stem cell transplant (SCT)(at any time) had significantly longer median OS. Although SCT at any time improved OS in the de novo myelodysplastic neoplasm group, the choice of the initial treatment with intensive chemotherapy, hypomethylating agents, or SCT did not significantly alter OS. Median OS was shorter in the pediatric group (<18 years old) and longer for those with isolated deletion of 5q or TET2 mutation, but these were not significant findings. Median OS was significantly shorter in those with monosomy 7 or 7q deletion and those with therapy-related myelodysplastic neoplasms. These findings build on previously reported findings and encourage the use of SCT along with molecular and cytogenetic analysis.

摘要

骨髓增生异常综合征,或骨髓增生异常肿瘤,在儿科、青少年和青年成人(AYA)患者中较为罕见。需要更多的文献来突出亚群的生存或治疗抵抗趋势,以改善治疗。在这里,我们报告了 2000 年至 2022 年的单中心回顾性分析,包括分子和细胞遗传学数据。使用仅在成人中报告的 IPSS-R 和 IPSS-M,并排除骨髓衰竭综合征患者,我们分析了 119 例儿科和 AYA 骨髓增生异常肿瘤患者。治疗相关的骨髓增生异常肿瘤存在于 36%的患者中,31%的患者发展为急性髓系白血病。整个队列的 5 年总生存率(OS)为 45%。与年轻成人和老年成人不同,儿科患者的突变并不常见。任何时间进行干细胞移植(SCT)的患者中位 OS 明显延长。尽管任何时间的 SCT 都改善了初发性骨髓增生异常肿瘤组的 OS,但初始治疗选择强化化疗、低甲基化药物或 SCT 并未显著改变 OS。儿科组(<18 岁)的中位 OS 较短,而孤立性 5q 缺失或 TET2 突变的患者中位 OS 较长,但这些都不是显著发现。中位 OS 在单体 7 或 7q 缺失和治疗相关骨髓增生异常肿瘤患者中明显缩短。这些发现建立在以前报道的发现基础上,并鼓励使用 SCT 以及分子和细胞遗传学分析。

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