Michiels Jan Jacques, Tevet Mihaela, Trifa Adrian, Niculescu-Mizil Emilia, Lupu Anca, Vladareanu Ana Maria, Bumbea Horia, Ilea Anca, Dobrea Camelia, Georgescu Daniela, Patrinoiu Oana, Popescu Mihaela, Murat Meilin, Dragan Cornel, Mihai Felicia, Zurac Sabina, Angelescu Silvana, Iova Anamaria, Popa Alina, Gogulescu Rodica, Popov Violeta
International Hematology and Bloodcoagulation Research Center, Goodheart Institute and Foundation in Nature Medicine, and International Collaboration and Research on Myeloproliferative Neoplasms: ICAR.MPN, Rotterdam, The Netherlands.
Department of Hematology, Colentina Clinical Hospital, Bucharest, Romania.
Maedica (Bucur). 2016 Mar;11(1):5-25.
The 2016 WHO-CMP classification proposal defines a broad spectrum of JAK2 V617F mutated MPN phenotypes: normocellular ET, hypercellular ET due to increased erythropoiesis (prodromal PV), hypercellular ET with megakaryocytic-granulocytic myeloproliferation and splenomegaly (EMGM or masked PV), erythrocythemic PV, early and overt classical PV, advanced PV with MF and post-PV MF. ET heterozygous for the JAK2 V617F mutation is associated with low JAK2 mutation load and normal life expectance. PV patients are hetero-homozygous versus homozygous for the JAK2 V617F mutation in their early versus advanced stages with increasing JAK2 mutation load from less than 50% to 100% and increase of MPN disease burden during life long follow-up in terms of symptomatic splenomegaly, constitutional symptoms, bone marrow hypercellularity and secondary MF. Pretreatment bone marrow biopsy in prefibrotic MPNs is of diagnostic and prognostic importance. JAK2 exon 12 mutated MPN is a distinct benign early stage PV. CALR mutated hypercellular thrombocythemia show distinct PMGM bone marrow characteristics of clustered larged immature dysmorphic megakaryocytes with bulky (bulbous) hyperchromatic nuclei, which are not seen in JAK2 mutated ET and PV. MPL mutated normocellular thrombocythemia is featured by clustered giant megakaryocytes with hyperlobulated stag-horn-like nuclei without features of PV in blood and bone marrow. Myeloproliferative disease burden in each of the JAK2, CALR and MPL MPNs is best reflected by the degree of anemia, splenomegaly, mutation allele burden, bone marrow cellularity and myelofibrosis.
2016年世界卫生组织慢性髓性肿瘤(WHO-CMP)分类提案定义了一系列广泛的JAK2 V617F突变的骨髓增殖性肿瘤(MPN)表型:正常细胞性原发性血小板增多症(ET)、因红细胞生成增加导致的高细胞性ET(前驱性真性红细胞增多症(PV))、伴有巨核细胞-粒细胞骨髓增殖和脾肿大的高细胞性ET(EMGM或隐匿性PV)、红细胞增多性PV、早期和明显的经典PV、伴有骨髓纤维化(MF)的晚期PV以及PV后MF。JAK2 V617F突变杂合的ET与低JAK2突变负荷和正常预期寿命相关。PV患者在疾病早期为JAK2 V617F突变的杂合子/纯合子,而在晚期为纯合子,JAK2突变负荷从低于50%增加到100%,并且在长期随访期间,MPN疾病负担在症状性脾肿大、全身症状、骨髓细胞增多和继发性MF方面增加。纤维化前MPN的预处理骨髓活检具有诊断和预后意义。JAK2外显子12突变的MPN是一种独特的良性早期PV。CALR突变的高细胞性血小板增多症表现出明显的PMGM骨髓特征,即聚集的大的未成熟畸形巨核细胞,其核大(球根状)且染色质深,这在JAK2突变的ET和PV中未见。MPL突变的正常细胞性血小板增多症的特征是聚集的巨大巨核细胞,其核呈多分叶状鹿角样,血液和骨髓中无PV特征。JAK2、CALR和MPL MPNs中每一种的骨髓增殖性疾病负担最好通过贫血程度、脾肿大、突变等位基因负担、骨髓细胞增多和骨髓纤维化来反映。