Department of Hematology and Oncology, Shanghai Children's Medical Center, School of Medicine, Shanghai Jiao Tong University, Dongfang Road 1678, Shanghai, 200127, China.
Eur J Pediatr. 2020 Mar;179(3):463-472. doi: 10.1007/s00431-019-03468-8. Epub 2019 Dec 5.
Juvenile myelomonocytic leukemia (JMML) is a heterogeneous childhood leukemia. The management of patients with JMML requires accurate assessment of genetic and clinical features to help in patient risk stratification. This study aimed to investigate the association between genomic alterations and prognosis in children with JMML. Genomic DNA was extracted from a total of 93 patients with JMML for targeted sequencing. Univariable and multivariable analysis were used to evaluate the correlation between gene mutations and prognosis of the patients. Patients with PTPN11 mutation exhibited significantly lower event-free survival (EFS) compared with non-PTPN11 mutations (P = 0.005). Patients without or with one somatic alteration at diagnosis showed significantly better prognosis in comparison with those with more than two alterations (P = 0.009). PTPN11 mutation with additional alterations showed significantly the poorest outcome in comparison with those with only one non-PTPN11 mutation, only one PTPN11 mutation, and combined mutations without PTPN11, respectively (P < 0.0001).Conclusion: Both PTPN11 mutation and the number of somatic alterations detected at diagnosis are likely to be the major determinant of outcome in JMML. The subgroup of patients with PTPN11 mutation showed the shortest survival which was even worsened when a secondary mutation was present.
儿童早幼粒细胞单核细胞白血病(JMML)是一种异质性儿童白血病。JMML 患者的管理需要准确评估遗传和临床特征,以帮助进行患者风险分层。本研究旨在探讨 JMML 患儿中基因组改变与预后的关系。从 93 例 JMML 患者中提取基因组 DNA 进行靶向测序。采用单变量和多变量分析评估基因突变与患者预后的相关性。与非 PTPN11 突变相比,PTPN11 突变患者的无事件生存(EFS)显著降低(P=0.005)。与存在两个以上改变的患者相比,诊断时无改变或仅有一个体细胞改变的患者预后明显更好(P=0.009)。与仅有一个非 PTPN11 突变、仅有一个 PTPN11 突变以及无 PTPN11 的联合突变相比,同时存在 PTPN11 突变和其他改变的患者预后明显更差(P<0.0001)。结论:PTPN11 突变和诊断时检测到的体细胞改变数量均可能是 JMML 结局的主要决定因素。PTPN11 突变患者亚组的生存最短,当存在继发突变时甚至更差。