Clinic for Paediatric and Adolescent Medicine, The Juliane Marie Centre, The University Hospital Rigshospitalet, Copenhagen.
Br J Haematol. 2012 May;157(4):476-82. doi: 10.1111/j.1365-2141.2012.09083.x. Epub 2012 Mar 7.
The chromosomal translocation t(12;21) resulting in the ETV6/RUNX1 fusion gene is the most frequent structural cytogenetic abnormality among patients with childhood acute lymphoblastic leukaemia (ALL). We investigated 62 ETV6/RUNX1-positive childhood ALL patients by single nucleotide polymorphism array to explore acquired copy number alterations (CNAs) at diagnosis. The mean number of CNAs was 2·82 (range 0-14). Concordance with available G-band karyotyping and comparative genomic hybridization was 93%. Based on three major protein-protein complexes disrupted by these CNAs, patients could be categorized into four distinct subgroups, defined by different underlying biological mechanisms relevant to the aetiology of childhood ALL. When recurrent CNAs were evaluated by an oncogenetic tree analysis classifying their sequential order, the most common genetic aberrations (deletions of 6q, 9p, 13q and X, and gains of 10 and 21) seemed independent of each other. Finally, we identified the most common regions with recurrent gains and losses, which comprise microRNA clusters with known oncogenic or tumour-suppressive roles. The present study sheds further light on the genetic diversity of ETV6/RUNX1-positive childhood ALL, which may be important for understanding poor responses among this otherwise highly curable subset of ALL and lead to novel targeted treatment strategies.
导致 ETV6/RUNX1 融合基因的染色体易位 t(12;21)是儿童急性淋巴细胞白血病 (ALL) 患者中最常见的结构性细胞遗传学异常。我们通过单核苷酸多态性微阵列分析了 62 例 ETV6/RUNX1 阳性的儿童 ALL 患者,以探讨初诊时获得性拷贝数改变 (CNA)。CNA 的平均数量为 2.82(范围 0-14)。与现有 G 带核型分析和比较基因组杂交的一致性为 93%。基于这些 CNA 破坏的三个主要蛋白质-蛋白质复合物,患者可以分为四个不同的亚组,这些亚组由与儿童 ALL 病因相关的不同潜在生物学机制定义。当通过分类其顺序的致癌基因树分析评估反复出现的 CNA 时,最常见的遗传异常(6q、9p、13q 和 X 的缺失以及 10 和 21 的获得)似乎彼此独立。最后,我们确定了最常见的反复增益和缺失区域,其中包括具有已知致癌或肿瘤抑制作用的 microRNA 簇。本研究进一步阐明了 ETV6/RUNX1 阳性儿童 ALL 的遗传多样性,这对于理解该高度可治愈的 ALL 亚组中不良反应可能很重要,并导致新的靶向治疗策略。