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真性红细胞增多症或原发性血小板增多症患者中纯合子JAK2 617V>F突变的临床特征。

Clinical profile of homozygous JAK2 617V>F mutation in patients with polycythemia vera or essential thrombocythemia.

作者信息

Vannucchi Alessandro M, Antonioli Elisabetta, Guglielmelli Paola, Rambaldi Alessandro, Barosi Giovanni, Marchioli Roberto, Marfisi Rosa Maria, Finazzi Guido, Guerini Vittoria, Fabris Fabrizio, Randi Maria Luigia, De Stefano Valerio, Caberlon Sabrina, Tafuri Agostino, Ruggeri Marco, Specchia Giorgina, Liso Vincenzo, Rossi Edoardo, Pogliani Enrico, Gugliotta Luigi, Bosi Alberto, Barbui Tiziano

机构信息

Department of Hematology, University of Florence, 50134 Florence, Italy.

出版信息

Blood. 2007 Aug 1;110(3):840-6. doi: 10.1182/blood-2006-12-064287. Epub 2007 Mar 22.

Abstract

JAK2 617V>F mutation occurs in a homozygous state in 25% to 30% of patients with polycythemia vera (PV) and 2% to 4% with essential thrombocythemia (ET). Whether homozygosity associates with distinct clinical phenotypes is still under debate. This retrospective multicenter study considered 118 JAK2 617V>F homozygous patients (104 PV, 14 ET) whose clinical characteristics were compared with those of 587 heterozygous and 257 wild-type patients. Irrespective of their clinical diagnosis, homozygous patients were older, displayed a higher leukocyte count and hematocrit value at diagnosis, and presented larger spleen volume. Aquagenic pruritus was significantly more common among homozygous PV patients. JAK2 617V>F homozygosity associated with more frequent evolution into secondary myelofibrosis in both PV and ET. After adjustment for sex, age, leukocyte count, and previous thrombosis in a multivariate analysis, homozygous ET patients displayed a significantly higher risk of cardiovascular events (hazard ratio [HR] 3.97, 95% confidence interval [CI] 1.34-11.7; P = .013) than wild-type (HR = 1.0) or heterozygous patients (HR = 1.49). No significant association of JAK2 617V>F homozygosity with thrombosis risk was observed in PV. Finally, JAK2 617V>F homozygous patients were more likely to receive chemotherapy for control of disease. We conclude that JAK2 617V>F homozygosity identifies PV or ET patients with a more symptomatic myeloproliferative disorder and is associated with a higher risk of major cardiovascular events in patients with ET.

摘要

在25%至30%的真性红细胞增多症(PV)患者以及2%至4%的原发性血小板增多症(ET)患者中,JAK2 617V>F突变以纯合状态出现。纯合性是否与不同的临床表型相关仍存在争议。这项回顾性多中心研究纳入了118例JAK2 617V>F纯合患者(104例PV患者,14例ET患者),并将其临床特征与587例杂合患者和257例野生型患者进行了比较。无论临床诊断如何,纯合患者年龄更大,诊断时白细胞计数和血细胞比容值更高,脾脏体积更大。在纯合PV患者中,水诱发性瘙痒明显更为常见。JAK2 617V>F纯合性与PV和ET患者更频繁地演变为继发性骨髓纤维化相关。在多变量分析中对性别、年龄、白细胞计数和既往血栓形成进行校正后,纯合ET患者发生心血管事件的风险(风险比[HR] 3.97,95%置信区间[CI] 1.34 - 11.7;P = 0.013)显著高于野生型患者(HR = 1.0)或杂合患者(HR = 1.49)。在PV患者中未观察到JAK2 617V>F纯合性与血栓形成风险之间存在显著关联。最后,JAK2 617V>F纯合患者更有可能接受化疗以控制疾病。我们得出结论,JAK2 617V>F纯合性可识别出具有更具症状性骨髓增殖性疾病的PV或ET患者,并且与ET患者发生主要心血管事件的较高风险相关。

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