Division of Hematology/Oncology, Department of Pediatrics, University of Alabama at Birmingham, Birmingham, AL.
Dana-Farber/Boston Children's Hospital Cancer and Blood Disorders Center, Boston, MA; and.
Blood Adv. 2021 Aug 24;5(16):3199-3202. doi: 10.1182/bloodadvances.2021004653.
Germline RUNX1 mutations underlie a syndrome, RUNX1-familial platelet disorder (RUNX1-FPD), characterized by bleeding symptoms that result from quantitative and/or qualitative defect in platelets and a significantly increased risk for developing hematologic malignancies. Myeloid neoplasms are the most commonly diagnosed hematologic malignancies, followed by lymphoid malignancies of T-cell origin. Here, we describe the first 2 cases of B-cell acute lymphoblastic leukemia (B-ALL) in patients with confirmed germline RUNX1 mutations. While 1 of the patients had a known diagnosis of RUNX1-FPD with a RUNX1 p.P240Hfs mutation, the other was the index patient of a kindred with a novel RUNX1 variant, RUNX1 c.587C>T (p.T196I), noted on a targeted genetic testing of the B-ALL diagnostic sample. We discuss the clinical course, treatment approaches, and the outcome for the 2 patients. Additionally, we describe transient resolution of the mild thrombocytopenia and bleeding symptoms during therapy, as well as the finding of clonal hematopoiesis with a TET2 mutant clone in 1 of the patients. It is critical to consider testing for germline RUNX1 mutations in patients presenting with B-ALL who have a personal or family history of thrombocytopenia, bleeding symptoms, or RUNX1 variants identified on genetic testing at diagnosis.
胚系 RUNX1 突变导致 RUNX1 家族性血小板疾病(RUNX1-FPD),其特征为血小板数量和/或质量缺陷导致的出血症状,以及发生血液系统恶性肿瘤的风险显著增加。髓系肿瘤是最常见的血液系统恶性肿瘤,其次是 T 细胞来源的淋巴母细胞性肿瘤。在此,我们描述了 2 例经证实存在胚系 RUNX1 突变的 B 细胞急性淋巴细胞白血病(B-ALL)患者。其中 1 例患者已知存在 RUNX1-FPD,携带 RUNX1 p.P240Hfs 突变,另 1 例是家族性病例的先证者,携带新的 RUNX1 变异,RUNX1 c.587C>T(p.T196I),在 B-ALL 诊断样本的靶向基因检测中发现。我们讨论了这 2 例患者的临床过程、治疗方法和结局。此外,我们描述了在治疗过程中,这 2 例患者的轻度血小板减少症和出血症状短暂缓解,以及其中 1 例患者存在克隆性造血,携带 TET2 突变克隆。在存在血小板减少症、出血症状或 RUNX1 变异家族史或诊断时在基因检测中发现 RUNX1 变异的 B-ALL 患者中,考虑进行胚系 RUNX1 突变检测至关重要。