Alvarez-Larrán A, Cervantes F, Bellosillo B, Giralt M, Juliá A, Hernández-Boluda J C, Bosch A, Hernández-Nieto L, Clapés V, Burgaleta C, Salvador C, Arellano-Rodrigo E, Colomer D, Besses C
Department of Hematology, Hospital del Mar, Barcelona, Spain.
Leukemia. 2007 Jun;21(6):1218-23. doi: 10.1038/sj.leu.2404693. Epub 2007 Apr 12.
The frequency of vascular events and evolution to myelofibrosis (MF) in young individuals with essential thrombocythemia (ET) is not well known. The incidence and predisposing factors to such complications was studied in 126 subjects diagnosed with ET at a median age of 31 years (range: 5-40). Overall survival and probability of survival free of thrombosis, bleeding and MF were analyzed by the Kaplan-Meier method and the presence of the Janus Kinase 2 (JAK2) V617F mutation correlated with the appearance of such complications. The JAK2 mutation (present in 43% of patients) was associated with higher hemoglobin (Hb) (P<0.001) and lower platelets at diagnosis. With a median follow-up of 10 years (range: 4-25), 31 thrombotic events were registered (incidence rate: 2.2 thromboses/100 patients/year). When compared with the general population, young ET patients showed a significant increase in stroke (odds ratio 50, 95% CI: 21.5-115) and venous thromboses (odds ratio 5.3, 95% CI: 3.9-10.6). Thrombosis-free survival was 84% at 10 years, with tobacco use being associated with higher risk of thrombosis. Actuarial freedom from evolution to MF was 97% at 10 years. In conclusion, young ET patients have thrombotic events, especially stroke and venous thrombosis, more frequently than generally considered, whereas they rarely transform to MF.
年轻原发性血小板增多症(ET)患者发生血管事件及进展为骨髓纤维化(MF)的频率尚不清楚。本研究对126例诊断为ET的患者(中位年龄31岁,范围5 - 40岁)的此类并发症的发生率及易感因素进行了研究。采用Kaplan-Meier法分析总生存率以及无血栓形成、出血和MF的生存概率,并分析Janus激酶2(JAK2)V617F突变的存在与这些并发症出现的相关性。JAK2突变(43%的患者存在)与诊断时较高的血红蛋白(Hb)水平(P<0.001)及较低的血小板计数相关。中位随访10年(范围4 - 25年),共记录到31例血栓事件(发生率:2.2次血栓/100例患者/年)。与普通人群相比,年轻ET患者的中风(优势比50,95%置信区间:21.5 - 115)和静脉血栓形成(优势比5.3,95%置信区间:3.9 - 10.6)显著增加。10年时无血栓生存为84%,吸烟与血栓形成风险较高相关。10年时进展为MF的精算无进展生存率为97%。总之,年轻ET患者发生血栓事件,尤其是中风和静脉血栓形成的频率高于一般认知,而他们很少转变为MF。