Yıldız Jale, Batgi Hikmettullah
Department of Hematology, Yıldırım Beyazıt University, Yenimahalle Training and Research Hospital, Ankara, TUR.
Department of Hematology, Ankara Training and Research Hospital, Ankara, TUR.
Cureus. 2022 Mar 15;14(3):e23171. doi: 10.7759/cureus.23171. eCollection 2022 Mar.
Introduction Essential thrombocythemia (ET) is one of the chronic myeloproliferative neoplasms. While Janus kinase 2 (JAK2) V617F mutation is defined in more than half of the patients with ET, calreticulin (CALR) or myeloproliferative leukemia virus oncogene (MPL) mutations are encountered more rarely. The discovery of the JAK2 V617F mutation in 2005, followed by the recognition of MPL and CALR mutations, brought up the idea of subdividing ET according to the mutation status. Our aim in this study is to investigate whether genetic mutations detected in patients diagnosed with ET cause a different clinical phenotype compared to triple-negative ET. Methods This retrospective study was conducted by evaluating the patients who were followed up with the diagnosis of ET in the hematology clinic of two tertiary centers in Turkey between 2009 and 2021. Patients with negative JAK2, CALR, and MPL mutations and meeting the diagnostic criteria for ET were defined as triple-negative ET. The patients were divided into two groups as triple-negative ET and mutation-positive ET according to the presence of a mutation. It was investigated whether there was a difference between these two groups in terms of demographic, laboratory, and clinical characteristics. Results A total of 109 patients were included in the study. The mean age of these patients was 54 (18-91) years and 85 (78%) patients were females. A total of 48 patients (44.0%) had JAK2 mutation, six (5.5%) had CALR mutation, and one (0.9%) had MPL mutation. It was observed that there was a significant difference between the two groups in terms of gender, mean age, and hemoglobin value. While 87% of patients with triple-negative ET were females, this rate was 69% in patients with mutation-positive ET (p = 0.036). The mean age was 41.8 years in triple-negative ET and 67.1 years in patients with mutation-positive ET (p = 0.0001). While the mean hemoglobin value was 12.9 g/dl in patients with triple-negative ET, it was 14.4 g/dl in patients with mutation-positive ET (p = 0.0001). Conclusion It has been observed that ET with JAK2, CALR, or MPL mutations may have different phenotypic features compared to triple-negative ET, resulting in a clinical condition consisting of older patients with a higher erythrocyte count.
引言
原发性血小板增多症(ET)是慢性骨髓增殖性肿瘤之一。虽然超过半数的ET患者存在Janus激酶2(JAK2)V617F突变,但钙网蛋白(CALR)或骨髓增殖性白血病病毒癌基因(MPL)突变则较为少见。2005年发现JAK2 V617F突变,随后又认识到MPL和CALR突变,这引发了根据突变状态对ET进行细分的想法。本研究的目的是调查诊断为ET的患者中检测到的基因突变与三阴性ET相比是否会导致不同的临床表型。
方法
本回顾性研究通过评估2009年至2021年期间在土耳其两个三级中心血液科门诊随访诊断为ET的患者进行。JAK2、CALR和MPL突变均为阴性且符合ET诊断标准的患者被定义为三阴性ET。根据是否存在突变将患者分为三阴性ET和突变阳性ET两组。研究这两组在人口统计学、实验室检查和临床特征方面是否存在差异。
结果
本研究共纳入109例患者。这些患者的平均年龄为54(18 - 91)岁,85例(78%)为女性。共有48例患者(44.0%)存在JAK2突变,6例(5.5%)存在CALR突变,1例(0.9%)存在MPL突变。观察到两组在性别、平均年龄和血红蛋白值方面存在显著差异。三阴性ET患者中87%为女性,而突变阳性ET患者中这一比例为69%(p = 0.036)。三阴性ET患者的平均年龄为41.8岁,突变阳性ET患者为67.1岁(p = 0.0001)。三阴性ET患者的平均血红蛋白值为12.9 g/dl,突变阳性ET患者为14.4 g/dl(p = 0.0001)。
结论
观察到与三阴性ET相比,存在JAK2、CALR或MPL突变的ET可能具有不同的表型特征,导致临床情况为红细胞计数较高的老年患者。