Deuitch Natalie T, Kajdic Amra, Bresciani Erica, Horwitz Marshall S, Scott Hamish S, Craft Katie, Chong Shawn, Young David J, Godley Lucy A, Liu Paul P
Oncogenesis and Development Section, Translational and Functional Genomics Branch, National Human Genome Research Institute, NIH, Bethesda, MD, USA.
Laboratory Medicine & Pathology, University of Washington School of Medicine, Seattle, WA, USA.
BJC Rep. 2025 Mar 27;3(1):18. doi: 10.1038/s44276-024-00117-y.
Pathogenic/likely pathogenic (P/LP) germline variants in RUNX1 cause familial platelet disorder with associated myeloid malignancies (FPDMM), also known as RUNX1-Familial Platelet Disorder (RUNX1-FPD, or FPD), a condition characterized by qualitative and quantitative platelet defects and predisposition to hematopoietic malignancies. Here, we present follow up to a case of a woman with acute myeloid leukemia and lifelong thrombocytopenia which had previously been attributed to presumptive pathogenic (P) GATA2 missense variants. However, re-evaluation with updated molecular technology sensitive for detection of copy number variants (CNVs) led to the identification of a P deletion of exons 5-6 in RUNX1, which had been undetected when examined at first presentation. This case highlights the importance of comprehensive molecular evaluation and careful variant interpretation, especially regarding CNVs.
RUNX1基因中的致病性/可能致病性(P/LP)种系变异会导致伴有相关髓系恶性肿瘤的家族性血小板疾病(FPDMM),也称为RUNX1家族性血小板疾病(RUNX1-FPD,或FPD),其特征是血小板在质量和数量上存在缺陷,并易患造血系统恶性肿瘤。在此,我们报告了一例患有急性髓系白血病且终生血小板减少症的女性病例的随访情况,该病例此前被认为是由推测致病性(P)GATA2错义变异所致。然而,采用对检测拷贝数变异(CNV)敏感的更新分子技术进行重新评估后,发现RUNX1基因外显子5-6存在一个P缺失,该缺失在首次就诊时未被检测到。该病例凸显了全面分子评估和仔细变异解读的重要性,尤其是对于CNV。