Jankowska Anna M, Szpurka Hadrian, Tiu Ramon V, Makishima Hideki, Afable Manuel, Huh Jungwon, O'Keefe Christine L, Ganetzky Rebecca, McDevitt Michael A, Maciejewski Jaroslaw P
Department of Translational Hematology and Oncology Research, Taussig Cancer Institute, Cleveland Clinic, OH 44195, USA.
Blood. 2009 Jun 18;113(25):6403-10. doi: 10.1182/blood-2009-02-205690. Epub 2009 Apr 16.
Chromosomal abnormalities are frequent in myeloid malignancies, but in most cases of myelodysplasia (MDS) and myeloproliferative neoplasms (MPN), underlying pathogenic molecular lesions are unknown. We identified recurrent areas of somatic copy number-neutral loss of heterozygosity (LOH) and deletions of chromosome 4q24 in a large cohort of patients with myeloid malignancies including MDS and related mixed MDS/MPN syndromes using single nucleotide polymorphism arrays. We then investigated genes in the commonly affected area for mutations. When we sequenced TET2, we found homozygous and hemizygous mutations. Heterozygous and compound heterozygous mutations were found in patients with similar clinical phenotypes without LOH4q24. Clinical analysis showed most TET2 mutations were present in patients with MDS/MPN (58%), including CMML (6/17) or sAML (32%) evolved from MDS/MPN and typical MDS (10%), suggesting they may play a ubiquitous role in malignant evolution. TET2 mutations affected conserved domains and the N terminus. TET2 is widely expressed in hematopoietic cells but its function is unknown, and it lacks homology to other known genes. The frequency of mutations in this candidate myeloid regulatory gene suggests an important role in the pathogenesis of poor prognosis MDS/MPN and sAML and may act as a disease gene marker for these often cytogenetically normal disorders.
染色体异常在髓系恶性肿瘤中很常见,但在大多数骨髓增生异常综合征(MDS)和骨髓增殖性肿瘤(MPN)病例中,潜在的致病分子病变尚不清楚。我们使用单核苷酸多态性阵列,在包括MDS以及相关的MDS/MPN混合综合征在内的一大群髓系恶性肿瘤患者中,鉴定出了体细胞拷贝数中性杂合性缺失(LOH)的复发区域以及4号染色体q24区域的缺失。然后,我们研究了常见受累区域的基因是否存在突变。当我们对TET2进行测序时,发现了纯合和半合子突变。在没有4q24 LOH的具有相似临床表型的患者中发现了杂合和复合杂合突变。临床分析表明,大多数TET2突变存在于MDS/MPN患者中(58%),包括CMML(6/17)或由MDS/MPN演变而来的sAML(32%)以及典型的MDS(10%),这表明它们可能在恶性演变中起普遍作用。TET2突变影响保守结构域和N末端。TET2在造血细胞中广泛表达,但其功能尚不清楚,并且它与其他已知基因缺乏同源性。这个候选髓系调节基因的突变频率表明它在预后不良的MDS/MPN和sAML的发病机制中起重要作用,并且可能作为这些通常细胞遗传学正常的疾病的疾病基因标志物。