Genomics Unit, Gregorio Marañón General University Hospital, Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain.
Gregorio Marañón Health Research Institute (IiSGM), Madrid, Spain.
Mol Oncol. 2021 Sep;15(9):2273-2284. doi: 10.1002/1878-0261.12921. Epub 2021 Jul 16.
Myeloid neoplasms (MN) with germline predisposition (MNGP) are likely to be more common than currently appreciated. Many of the genes involved in MNGP are also recurrently mutated in sporadic MN. Therefore, routine analysis of gene panels by next-generation sequencing provides an effective approach to detect germline variants with clinical significance in patients with hematological malignancies. Gene panel sequencing was performed in 88 consecutive and five nonconsecutive patients with MN diagnosis. Disease-causing germline mutations in CEBPα, ASXL1, TP53, MPL, GATA2, DDX41, and ETV6 genes were identified in nine patients. Six out of the nine patients with germline variants had a strong family history. These patients presented great heterogeneity in the age of diagnosis and phenotypic characteristics. In our study, there were families in which all the affected members presented the same subtype of disease, whereas members of other families presented various disease phenotypes. This intrafamiliar heterogeneity suggests that the acquisition of particular somatic variants may drive the evolution of the disease. This approach enabled high-throughput detection of MNGP in patients with MN diagnosis, which is of great relevance for both the patients themselves and the asymptomatic mutation carriers within the family. It is crucial to make a proper diagnosis of these patients to provide them with the most suitable treatment, follow-up, and genetic counseling.
具有种系倾向的髓系肿瘤(MNGP)可能比目前认为的更为常见。许多与 MNGP 相关的基因也在散发性 MN 中经常发生突变。因此,通过下一代测序对基因进行常规分析为检测血液系统恶性肿瘤患者具有临床意义的种系变异提供了有效的方法。对 88 例连续和 5 例非连续的 MN 诊断患者进行了基因panel 测序。在 9 例患者中鉴定出 CEBPα、ASXL1、TP53、MPL、GATA2、DDX41 和 ETV6 基因的致病变异体。这 9 例种系变异患者中有 6 例具有强烈的家族史。这些患者在诊断时的年龄和表型特征上存在很大的异质性。在我们的研究中,有些家族的所有受影响成员都呈现出相同的疾病亚型,而其他家族的成员则呈现出不同的疾病表型。这种家族内异质性表明,特定的体细胞变异的获得可能会推动疾病的发展。这种方法能够在 MN 诊断患者中实现 MNGP 的高通量检测,这对于患者本身以及家族中的无症状突变携带者都具有重要意义。对这些患者进行正确的诊断对于为他们提供最合适的治疗、随访和遗传咨询至关重要。