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基于人群的285例儿童T细胞急性淋巴细胞白血病连续系列的临床和细胞遗传学特征:罕见的T细胞受体基因重排与不良预后相关。

Clinical and cytogenetic features of a population-based consecutive series of 285 pediatric T-cell acute lymphoblastic leukemias: rare T-cell receptor gene rearrangements are associated with poor outcome.

作者信息

Karrman Kristina, Forestier Erik, Heyman Mats, Andersen Mette K, Autio Kirsi, Blennow Elisabeth, Borgström Georg, Ehrencrona Hans, Golovleva Irina, Heim Sverre, Heinonen Kristiina, Hovland Randi, Johannsson Johann H, Kerndrup Gitte, Nordgren Ann, Palmqvist Lars, Johansson Bertil

机构信息

Department of Clinical Genetics, Lund University Hospital, Lund, Sweden.

出版信息

Genes Chromosomes Cancer. 2009 Sep;48(9):795-805. doi: 10.1002/gcc.20684.

Abstract

Clinical characteristics and cytogenetic aberrations were ascertained and reviewed in a population-based consecutive series of 285 pediatric T-cell acute lymphoblastic leukemias (T-ALLs) diagnosed between 1992 and 2006 in the Nordic countries. Informative karyotypic results were obtained in 249 (87%) cases, of which 119 (48%) were cytogenetically abnormal. Most (62%) of the aberrant T-ALLs were pseudodiploid. Structural changes were more common than numerical ones; 86% displayed at least one structural abnormality and 41% at least one numerical anomaly. The most frequent abnormalities were T-cell receptor (TCR) gene rearrangements (20%) [TCR;11p13 (10%), TCR;10q24 (3%), TCR;other (8%)], del(9p) (17%), +8 (14%), del(6q) (12%), and 11q23 rearrangements (6%). The TCR;other group comprised the rare rearrangements t(X;14)(p11;q11), t(X;7)(q22;q34), t(1;14)(p32;q11), ins(14;5)(q11;q?q?), inv(7)(p15q34), t(8;14)(q24;q11), t(7;11)(q34;p15), and t(12;14)(p13;q11). The clinical characteristics of this Nordic patient cohort agreed well with previous larger series, with a median age of 9.0 years, male predominance (male/female ratio 3.1), median white blood cell (WBC) count of 66.5 x 10(9)/l, and a high incidence of mediastinal mass and central nervous system involvement (59% and 9.5%, respectively). These features did not differ significantly among the various genetic subgroups. 5-year event-free survival (EFS) and overall survival for all patients were 0.61 (+/-0.03) and 0.67 (+/-0.03), respectively. In a multivariate analysis, two factors affected negatively the EFS, namely a WBC count of > or =200 x 10(9)/l (P < 0.001) and the presence of rare TCR rearrangements (P = 0.001). In conclusion, in this large series of childhood T-ALLs from the Nordic countries, the cytogenetic findings were not associated with risk of therapy failure with the exception of the TCR;other group. However, further prospective and collaborative investigations of this genetically heterogeneous entity are needed to confirm these results.

摘要

对1992年至2006年期间在北欧国家诊断出的285例儿童T细胞急性淋巴细胞白血病(T-ALL)进行了基于人群的连续系列研究,确定并回顾了其临床特征和细胞遗传学异常情况。249例(87%)病例获得了有信息价值的核型结果,其中119例(48%)细胞遗传学异常。大多数(62%)异常T-ALL为假二倍体。结构改变比数目改变更常见;86%显示至少一种结构异常,41%显示至少一种数目异常。最常见的异常是T细胞受体(TCR)基因重排(20%)[TCR;11p13(10%),TCR;10q24(3%),TCR;其他(8%)],del(9p)(17%),+8(14%),del(6q)(12%),以及11q23重排(6%)。TCR;其他组包括罕见重排t(X;14)(p11;q11)、t(X;7)(q22;q34)、t(1;14)(p32;q11)、ins(14;5)(q11;q?q?)、inv(7)(p15q34)、t(8;14)(q24;q11)、t(7;11)(q34;p15)和t(12;14)(p13;q11)。该北欧患者队列的临床特征与之前更大系列的研究结果非常一致,中位年龄为9.0岁,男性占优势(男/女比例为3.1),中位白细胞(WBC)计数为66.5×10⁹/L,纵隔肿块和中枢神经系统受累的发生率较高(分别为59%和9.5%)。这些特征在不同遗传亚组之间没有显著差异。所有患者的5年无事件生存率(EFS)和总生存率分别为0.61(±0.03)和0.67(±0.03)。在多变量分析中,有两个因素对EFS有负面影响,即WBC计数≥200×10⁹/L(P<0.001)和存在罕见的TCR重排(P = 0.001)。总之,在这一来自北欧国家的大量儿童T-ALL系列研究中,除了TCR;其他组外,细胞遗传学结果与治疗失败风险无关。然而,需要对这个基因异质性实体进行进一步的前瞻性和协作性研究以证实这些结果。

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