Combination of myeloproliferative neoplasm driver gene activation with mutations of splice factor or epigenetic modifier genes increases risk of rapid blastic progression

Eur J Haematol. 2021 Apr;106(4):520-528. doi: 10.1111/ejh.13579. Epub 2021 Feb 1.

Abstract

Objectives: Myeloproliferative neoplasms (MPN) comprising polycythemia vera (PV), essential thrombocythemia (ET) and primary myelofibrosis (PMF) follow a bi-phasic course of disease with fibrotic and/or blastic progression. At presentation in the chronic phase, currently there are only insufficient tools to predict the risk of progression in individual cases.

Methods: In this study, chronic phase MPN (16 PMF, 11 PV, and 11 MPN unclassified) with blastic transformation during course of disease (n = 38, median follow-up 5.3 years) were analyzed by high-throughput sequencing. MPN cases with a comparable follow-up period and without evidence of blast increase served as control (n = 63, median follow-up 5.8 years).

Results: Frequent ARCH/CHIP-associated mutations (TET2, ASXL1, DNMT3A) found at presentation were not significantly associated with blastic transformation. By contrast, mutations of SRSF2, U2AF1, and IDH1/2 at first presentation were frequently observed in the progression cohort (13/38, 34.2%) and were completely missing in the control group without blast transformation during follow-up (P = .0007 for SRSF2; P = .0063 for U2AF1 and IDH1/2).

Conclusion: Unlike frequent ARCH/CHIP alterations (TET2, ASXL1, DNMT3A), mutations in SRSF2, IDH1/2, and U2AF1 when manifest already at first presentation provide an independent risk factor for rapid blast transformation of MPN.

Keywords: biomarker; genetic risk stratification; leukemic transformation; myeloproliferative neoplasms.

MeSH terms

  • Aged
  • Blast Crisis
  • Child, Preschool
  • Disease Progression
  • Epigenesis, Genetic*
  • Female
  • Genes, Modifier*
  • Genetic Predisposition to Disease
  • Humans
  • Infant
  • Male
  • Middle Aged
  • Mutation*
  • Myeloproliferative Disorders / genetics*
  • Myeloproliferative Disorders / pathology*
  • Oncogenes*
  • RNA Splicing Factors / genetics*
  • Serine-Arginine Splicing Factors
  • Splicing Factor U2AF / genetics
  • Transcriptional Activation*

Substances

  • RNA Splicing Factors
  • Splicing Factor U2AF
  • U2AF1 protein, human
  • SRSF2 protein, human
  • Serine-Arginine Splicing Factors