Division of Hematology and Oncology, Department of Medicine, Chang Gung Memorial Hospital, Chiayi, 613, Taiwan.
College of Medicine, Chang Gung University, Taoyuan, 333, Taiwan.
Ann Hematol. 2024 Jun;103(6):1947-1965. doi: 10.1007/s00277-024-05754-4. Epub 2024 Apr 23.
Janus kinase 2 (JAK2) V617F mutation is present in most patients with polycythemia vera (PV). One persistently puzzling aspect unresolved is the association between JAK2V617F allele burden (also known as variant allele frequency) and the relevant clinical characteristics. Numerous studies have reported associations between allele burden and both hematologic and clinical features. While there are strong indications linking high allele burden in PV patients with symptoms and clinical characteristics, not all associations are definitive, and disparate and contradictory findings have been reported. Hence, this study aimed to synthesize existing data from the literature to better understand the association between JAK2V617F allele burden and relevant clinical correlates. Out of the 1,851 studies identified, 39 studies provided evidence related to the association between JAK2V617F allele burden and clinical correlates, and 21 studies were included in meta-analyses. Meta-analyses of correlation demonstrated that leucocyte and erythrocyte counts were significantly and positively correlated with JAK2V617F allele burden, whereas platelet count was not. Meta-analyses of standardized mean difference demonstrated that leucocyte and hematocrit were significantly higher in patients with higher JAK2V617F allele burden, whereas platelet count was significantly lower. Meta-analyses of odds ratio demonstrated that patients who had higher JAK2V617F allele burden had a significantly greater odds ratio for developing pruritus, splenomegaly, thrombosis, myelofibrosis, and acute myeloid leukemia. Our study integrates data from approximately 5,462 patients, contributing insights into the association between JAK2V617F allele burden and various hematological parameters, symptomatic manifestations, and complications. However, varied methods of data presentation and statistical analyses prevented the execution of high-quality meta-analyses.
Janus 激酶 2(JAK2)V617F 突变存在于大多数真性红细胞增多症(PV)患者中。一个尚未解决的令人费解的方面是 JAK2V617F 等位基因负担(也称为变异等位基因频率)与相关临床特征之间的关联。许多研究报告了等位基因负担与血液学和临床特征之间的关联。虽然有强有力的迹象表明 PV 患者的高等位基因负担与症状和临床特征有关,但并非所有关联都是明确的,并且已有报道称存在不一致和相互矛盾的发现。因此,本研究旨在综合文献中的现有数据,以更好地了解 JAK2V617F 等位基因负担与相关临床指标之间的关系。在确定的 1851 项研究中,有 39 项研究提供了与 JAK2V617F 等位基因负担与临床相关因素之间的关联相关的证据,其中 21 项研究被纳入荟萃分析。相关性的荟萃分析表明,白细胞和红细胞计数与 JAK2V617F 等位基因负担呈显著正相关,而血小板计数则不然。标准化均数差的荟萃分析表明,JAK2V617F 等位基因负担较高的患者白细胞和血细胞比容显著较高,而血小板计数显著较低。比值比的荟萃分析表明,JAK2V617F 等位基因负担较高的患者发生瘙痒、脾肿大、血栓形成、骨髓纤维化和急性髓系白血病的可能性显著增加。本研究整合了大约 5462 名患者的数据,深入了解了 JAK2V617F 等位基因负担与各种血液学参数、症状表现和并发症之间的关系。然而,数据呈现和统计分析方法的差异妨碍了高质量荟萃分析的执行。