• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

PTPN11 突变伴发其他体细胞改变提示少年骨髓单核细胞白血病不良预后:来自单中心的回顾性临床研究。

PTPN11 mutation with additional somatic alteration indicates unfavorable outcome in juvenile myelomonocytic leukemia: a retrospective clinical study from a single center.

机构信息

Department of Hematology and Oncology, Shanghai Children's Medical Center, School of Medicine, Shanghai Jiao Tong University, Dongfang Road 1678, Shanghai, 200127, China.

出版信息

Eur J Pediatr. 2020 Mar;179(3):463-472. doi: 10.1007/s00431-019-03468-8. Epub 2019 Dec 5.

DOI:10.1007/s00431-019-03468-8
PMID:31807902
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7028800/
Abstract

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.

摘要

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。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38dd/7028800/b2f26bb9fead/431_2019_3468_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38dd/7028800/34db39a5bf32/431_2019_3468_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38dd/7028800/b2f26bb9fead/431_2019_3468_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38dd/7028800/34db39a5bf32/431_2019_3468_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/38dd/7028800/b2f26bb9fead/431_2019_3468_Fig2_HTML.jpg

相似文献

1
PTPN11 mutation with additional somatic alteration indicates unfavorable outcome in juvenile myelomonocytic leukemia: a retrospective clinical study from a single center.PTPN11 突变伴发其他体细胞改变提示少年骨髓单核细胞白血病不良预后:来自单中心的回顾性临床研究。
Eur J Pediatr. 2020 Mar;179(3):463-472. doi: 10.1007/s00431-019-03468-8. Epub 2019 Dec 5.
2
[Analysis of Gene Mutation and Clinical Characteristics in 19 Children with Juvenile Myelomonocytic Leukemia].19例青少年粒单核细胞白血病患儿基因突变及临床特征分析
Zhongguo Shi Yan Xue Ye Xue Za Zhi. 2020 Dec;28(6):1811-1818. doi: 10.19746/j.cnki.issn.1009-2137.2020.06.004.
3
Correlation of clinical features with the mutational status of GM-CSF signaling pathway-related genes in juvenile myelomonocytic leukemia.青少年粒单核细胞白血病临床特征与GM-CSF信号通路相关基因突变状态的相关性
Pediatr Res. 2009 Mar;65(3):334-40. doi: 10.1203/PDR.0b013e3181961d2a.
4
Juvenile myelomonocytic leukaemia and Noonan syndrome.青少年型骨髓单核细胞白血病与努南综合征。
J Med Genet. 2014 Oct;51(10):689-97. doi: 10.1136/jmedgenet-2014-102611. Epub 2014 Aug 5.
5
Myeloid Dysregulation in a Human Induced Pluripotent Stem Cell Model of PTPN11-Associated Juvenile Myelomonocytic Leukemia.PTPN11相关青少年粒单核细胞白血病人诱导多能干细胞模型中的髓系失调
Cell Rep. 2015 Oct 20;13(3):504-515. doi: 10.1016/j.celrep.2015.09.019. Epub 2015 Oct 8.
6
Gene mutations in the Ras pathway and the prognostic implication in Korean patients with juvenile myelomonocytic leukemia.Ras 通路中的基因突变与韩国青少年骨髓单核细胞白血病患者的预后意义。
Ann Hematol. 2012 Apr;91(4):511-7. doi: 10.1007/s00277-011-1326-9. Epub 2011 Sep 8.
7
Juvenile myelomonocytic leukemia: who's the driver at the wheel?幼年型粒单核细胞白血病:谁是掌舵人?
Blood. 2019 Mar 7;133(10):1060-1070. doi: 10.1182/blood-2018-11-844688. Epub 2019 Jan 22.
8
[Gene mutations and clinical characteristics in children with juvenile myelomonocytic leukemia].[青少年型骨髓单核细胞白血病患儿的基因突变与临床特征]
Zhongguo Dang Dai Er Ke Za Zhi. 2015 Jan;17(1):1-5.
9
[Clinical features and prognosis of juvenile myelomonocytic leukemia: an analysis of 63 cases].青少年粒单核细胞白血病的临床特征与预后:63例分析
Zhongguo Dang Dai Er Ke Za Zhi. 2023 Mar 15;25(3):265-271. doi: 10.7499/j.issn.1008-8830.2209129.
10
Nonsyndromic Juvenile Myelomonocytic Leukemia With PTPN11 Mutation in a 9-Year-old Girl.一名9岁女孩患伴有PTPN11突变的非综合征性青少年骨髓单核细胞白血病。
J Pediatr Hematol Oncol. 2015 Aug;37(6):486-7. doi: 10.1097/MPH.0000000000000363.

引用本文的文献

1
hypomethylation drives oncogenic signaling and stratifies poor prognosis in juvenile myelomonocytic leukemia.低甲基化驱动致癌信号传导并导致青少年粒单核细胞白血病预后不良。
Transl Pediatr. 2025 Jul 31;14(7):1541-1552. doi: 10.21037/tp-2025-228. Epub 2025 Jul 28.
2
SFX-01 is therapeutic against myeloproliferative disorders caused by activating mutations in Shp2.SFX-01对由Shp2激活突变引起的骨髓增殖性疾病具有治疗作用。
EMBO Mol Med. 2025 Jul 10. doi: 10.1038/s44321-025-00267-7.
3
Research progress of additional pathogenic mutations in chronic neutrophilic leukemia.

本文引用的文献

1
Integrated molecular profiling of juvenile myelomonocytic leukemia.青少年髓单核细胞白血病的综合分子分析。
Blood. 2018 Apr 5;131(14):1576-1586. doi: 10.1182/blood-2017-07-798157. Epub 2018 Feb 2.
2
RAS-pathway mutation patterns define epigenetic subclasses in juvenile myelomonocytic leukemia.RAS 通路突变模式定义青少年骨髓单核细胞白血病的表观遗传亚型。
Nat Commun. 2017 Dec 19;8(1):2126. doi: 10.1038/s41467-017-02177-w.
3
Chronic Cigarette Smoke-Induced Epigenomic Changes Precede Sensitization of Bronchial Epithelial Cells to Single-Step Transformation by KRAS Mutations.
慢性中性粒细胞白血病中附加致病性突变的研究进展。
Ann Hematol. 2024 Aug;103(8):2591-2600. doi: 10.1007/s00277-023-05550-6. Epub 2023 Nov 22.
慢性香烟烟雾诱导的表观基因组变化先于支气管上皮细胞对KRAS突变单步转化的致敏作用。
Cancer Cell. 2017 Sep 11;32(3):360-376.e6. doi: 10.1016/j.ccell.2017.08.006.
4
Inhibition of the Gab2/PI3K/mTOR signaling ameliorates myeloid malignancy caused by Ptpn11 (Shp2) gain-of-function mutations.抑制Gab2/PI3K/mTOR信号传导可改善由Ptpn11(Shp2)功能获得性突变引起的髓系恶性肿瘤。
Leukemia. 2017 Jun;31(6):1415-1422. doi: 10.1038/leu.2016.326. Epub 2016 Nov 14.
5
The 2016 revision to the World Health Organization classification of myeloid neoplasms and acute leukemia.2016 年版世界卫生组织髓系肿瘤和急性白血病分类。
Blood. 2016 May 19;127(20):2391-405. doi: 10.1182/blood-2016-03-643544. Epub 2016 Apr 11.
6
Epigenetic instability at imprinting control regions in a Kras(G12D)-induced T-cell neoplasm.印记控制区的表观遗传不稳定性在 Kras(G12D)诱导的 T 细胞肿瘤中。
Epigenetics. 2015;10(12):1111-20. doi: 10.1080/15592294.2015.1110672.
7
How I treat juvenile myelomonocytic leukemia.我如何治疗青少年骨髓单核细胞白血病。
Blood. 2015 Feb 12;125(7):1083-90. doi: 10.1182/blood-2014-08-550483. Epub 2015 Jan 6.
8
Bedside to bench in juvenile myelomonocytic leukemia: insights into leukemogenesis from a rare pediatric leukemia.从一种罕见儿科白血病看少年骨髓单核细胞白血病的发病机制:从床边到实验台。
Blood. 2014 Oct 16;124(16):2487-97. doi: 10.1182/blood-2014-03-300319. Epub 2014 Aug 27.
9
A KRAS-directed transcriptional silencing pathway that mediates the CpG island methylator phenotype.一种介导CpG岛甲基化表型的KRAS定向转录沉默途径。
Elife. 2014 Mar 12;3:e02313. doi: 10.7554/eLife.02313.
10
Ras and Rac1, frequently mutated in melanomas, are activated by superoxide anion, modulate Dnmt1 level and are causally related to melanocyte malignant transformation.Ras和Rac1在黑色素瘤中经常发生突变,被超氧阴离子激活,调节Dnmt1水平,并与黑素细胞恶性转化有因果关系。
PLoS One. 2013 Dec 16;8(12):e81937. doi: 10.1371/journal.pone.0081937. eCollection 2013.