Ann Hematol. 2014 Dec;93(12):2029-36. doi: 10.1007/s00277-014-2151-8.
Calreticulin (CALR) mutations were recently identified in patients with essential thrombocythemia (ET) and primary myelofibrosis (PMF) devoid of JAK2 and MPL mutations. We evaluated the clinical, laboratory, and molecular features of a Taiwanese population of patients with ET. Among 147 ET patients, CALR mutations were detected in 33 (22.5 %), JAK2V617F in 94 (63.9 %), and MPL mutations in 4 (2.7 %). Sixteen (10.9 %) patients were negative for all three mutations (CALR, JAK2V617F, and MPL; triple negative). Interestingly, one patient with the type 2 CALR mutation also harbored a low allele burden (0.025 %) of JAK2V617F mutation. Furthermore, we found a novel CALR mutation, with the resultant protein sharing an identical amino acid sequence to the type 6 CALR mutant. Compared to those with JAK2 mutation, CALR-mutated ET patients were characterized by younger age, lower leukocyte count, higher platelet count, and decreased risk of thrombosis. CALR mutations had a favorable impact on thrombosis-free survival (TFS) for ET patients, whereas the respective TFS outcomes were similarly poorer in JAK2-mutated ET and PV patients. Multivariate analysis confirmed that younger age (<60 years), presence of CALR mutations, and a lower platelet count (<1,000 × 10(9)/L) were independently associated with a longer TFS in ET patients. The current study demonstrates that CALR mutations characterize a special group of ET patients with unique phenotypes that are not discrepant from those seen in Western countries.
钙网织蛋白(CALR)突变最近在无 JAK2 和 MPL 突变的原发性骨髓纤维化(PMF)和特发性血小板增多症(ET)患者中被发现。我们评估了台湾 ET 患者的临床、实验室和分子特征。在 147 名 ET 患者中,检测到 33 名(22.5%)患者存在 CALR 突变,94 名(63.9%)患者存在 JAK2V617F 突变,4 名(2.7%)患者存在 MPL 突变。16 名(10.9%)患者三种突变均为阴性(CALR、JAK2V617F 和 MPL;三阴性)。有趣的是,一名存在 2 型 CALR 突变的患者还存在 JAK2V617F 突变的低等位基因负担(0.025%)。此外,我们发现了一种新的 CALR 突变,其导致的蛋白质与 6 型 CALR 突变体具有相同的氨基酸序列。与 JAK2 突变患者相比,CALR 突变的 ET 患者具有更年轻的年龄、更低的白细胞计数、更高的血小板计数和降低的血栓形成风险。CALR 突变对 ET 患者的无血栓生存(TFS)有有利影响,而 JAK2 突变的 ET 和 PV 患者的 TFS 结果则类似更差。多变量分析证实,年龄较轻(<60 岁)、存在 CALR 突变和血小板计数较低(<1000×10(9)/L)与 ET 患者的 TFS 延长独立相关。本研究表明,CALR 突变可将 ET 患者分为具有独特表型的特殊亚组,与西方国家的表型并无不同。