Pre-fibrotic/early primary myelofibrosis vs. WHO-defined essential thrombocythemia: The impact of minor clinical diagnostic criteria on the outcome of the disease

Am J Hematol. 2017 Sep;92(9):885-891. doi: 10.1002/ajh.24788. Epub 2017 Jun 9.

Abstract

The 2016 revised WHO criteria for the diagnosis of pre-fibrotic/early primary myelofibrosis (pre-PMF) require at least one of the following four borderline expressed minor clinical criteria: anemia, leukocytosis, elevated lactate dehydrogenase and splenomegaly. In this study, we evaluated the relative frequency of these four criteria in a group of 170 pre-PMF patients and compared them to 225 ET cases. More than 91% of pre-PMF cases showed one or more of these features required for diagnosis, by contrast with only 48% of ET patients. According to clinical data the cumulative risk of progression to advanced/overt PMF in pre-PMF was 36.9% after 15 years. After fitting cox regression models to analyze the impact of the minor criteria on overall survival, only leukocytosis remained as a significant predictor of survival in both pre-PMF and ET. Molecular characterization showed differences in survival in pre-PMF but not ET, with CALR being a more favorable mutation than JAK2. The different outcome of pre-PMF versus ET and associated molecular genetic data supports the concept of two different entities, rather than a continuum of the same disease. Although slightly less than 50% of ET patients also show one or more minor clinical criteria, accurate distinction between ET and pre-PMF is possible by following an integrated approach including histomorphological diagnosis and presence of minor clinical criteria.

Publication types

  • Clinical Trial
  • Comparative Study
  • Multicenter Study

MeSH terms

  • Aged
  • Calreticulin / genetics
  • Disease-Free Survival
  • Female
  • Follow-Up Studies
  • Humans
  • Janus Kinase 2 / genetics
  • Male
  • Middle Aged
  • Mutation
  • Primary Myelofibrosis / diagnosis*
  • Primary Myelofibrosis / genetics
  • Primary Myelofibrosis / mortality*
  • Primary Myelofibrosis / therapy
  • Risk Factors
  • Survival Rate
  • Thrombocythemia, Essential / diagnosis*
  • Thrombocythemia, Essential / genetics
  • Thrombocythemia, Essential / mortality*
  • Thrombocythemia, Essential / therapy
  • World Health Organization

Substances

  • CALR protein, human
  • Calreticulin
  • JAK2 protein, human
  • Janus Kinase 2