Impact of genotype on leukaemic transformation in polycythaemia vera and essential thrombocythaemia

Br J Haematol. 2017 Sep;178(5):764-771. doi: 10.1111/bjh.14762. Epub 2017 May 23.

Abstract

The influence of driver mutations on leukaemic transformation was analysed in 1747 patients with polycythaemia vera or essential thrombocythaemia. With a median follow-up of 7·2 years, 349 patients died and 62 progressed to acute leukaemia or myelodysplastic syndrome. Taking death as a competing risk, CALR genotype was associated with a lower risk of transformation [subdistribution hazard ratio (SHR): 0·13, 95% confidence interval (CI): 0·2-0·9, P = 0·039], whereas JAK2 V617F showed borderline significance for higher risk (SHR: 2·05, 95% CI: 0·9-4·6, P = 0·09). Myelofibrotic transformation increased leukaemic risk, except in CALR-mutated patients. Next generation sequencing of 51 genes at the time of transformation showed additional mutations (median number: 3; range: 1-5) in 25 out of 29 (86%) assessable cases. Mutations (median: 1; range: 1-3) were detected in 67% of paired samples from the chronic phase. Leukaemia appeared in a JAK2 V617F negative clone in 17 (58%) cases, eleven of them being previously JAK2 V617F-positive. JAK2 V617F-mutated leukaemia was significantly associated with complex karyotype and acquisition of TP53 mutations, whereas EZH2 and RUNX1 mutations were more frequent in JAK2 V617F-negative leukaemia. Survival was longer in JAK2 V617F-unmutated leukaemia (343 days vs. 95 days, P = 0·003). In conclusion, CALR genotype is associated with a lower risk of leukaemic transformation. Leukaemia arising in a JAK2 V617F-negative clone is TP53 independent and shows better survival.

Keywords: essential thrombocythaemia; genotype; myeloid leukaemia; polycythaemia vera; prognostic factors.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Calreticulin / genetics
  • Cell Transformation, Neoplastic / genetics*
  • Child
  • Female
  • Follow-Up Studies
  • Genotype
  • Humans
  • Janus Kinase 2 / genetics
  • Kaplan-Meier Estimate
  • Leukemia, Myeloid / genetics*
  • Leukemia, Myeloid / mortality
  • Male
  • Middle Aged
  • Mutation
  • Myelodysplastic Syndromes / genetics*
  • Myelodysplastic Syndromes / mortality
  • Polycythemia Vera / genetics*
  • Polycythemia Vera / mortality
  • Prognosis
  • Risk Factors
  • Spain / epidemiology
  • Thrombocythemia, Essential / genetics*
  • Thrombocythemia, Essential / mortality
  • Young Adult

Substances

  • Calreticulin
  • JAK2 protein, human
  • Janus Kinase 2