Second primary malignancy in myelofibrosis patients treated with ruxolitinib

Br J Haematol. 2021 Apr;193(2):356-368. doi: 10.1111/bjh.17192. Epub 2020 Nov 21.

Abstract

Ruxolitinib (RUX), the first JAK1/JAK2 inhibitor approved for myelofibrosis (MF) therapy, has recently been associated with the occurrence of second primary malignancies (SPMs), mainly lymphomas and non-melanoma skin cancers (NMSCs). We analyzed the incidence, risk factors and outcome of SPMs in 700 MF patients treated with RUX in a real-world context. Median follow-up from starting RUX was 2·9 years. Overall, 80 (11·4%) patients developed 87 SPMs after RUX start. NMSCs were the most common SPMs (50·6% of the cases). Multivariate analysis demonstrated that male sex [hazard ratio (HR): 2·37, 95% confidence interval (95%CI): 1·22-4·60, P = 0·01] and thrombocytosis> 400 × 109 /l at RUX start (HR:1·98, 95%CI: 1·10-4·60, P = 0·02) were associated with increased risk for SPMs. Risk factors for NMSC alone were male sex (HR: 3·14, 95%CI: 1·24-7·92, P = 0·02) and duration of hydroxycarbamide and RUX therapy > 5 years (HR: 3·20, 95%CI: 1·17-8·75, P = 0·02 and HR: 2·93, 95%CI: 1·39-6·17, P = 0·005 respectively). In SPMs excluding NMSCs, male sex (HR: 2·41, 95%CI: 1·11-5·25, P = 0·03), platelet > 400 × 109 /l (HR: 3·30, 95%CI: 1·67-6·50, P = 0·001) and previous arterial thromboses (HR: 3·47, 95%CI: 1·48-8·14, P = 0·004) were shown to be associated with higher risk of SPMs. While it is reassuring that no aggressive lymphoma was documented, active skin surveillance is recommended in all patients and particularly after prolonged hydroxycaramide therapy; oncological screening should be triggered by thrombocytosis and arterial thrombosis, particularly in males.

Keywords: JAK inhibitors; myelofibrosis; ruxolitinib; second cancer; toxicity.

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Arteries / pathology
  • Case-Control Studies
  • Female
  • Follow-Up Studies
  • Humans
  • Incidence
  • Janus Kinase Inhibitors / administration & dosage
  • Janus Kinase Inhibitors / adverse effects*
  • Janus Kinase Inhibitors / therapeutic use
  • Janus Kinase Inhibitors / toxicity
  • Lymphoma / diagnosis
  • Lymphoma / epidemiology
  • Male
  • Middle Aged
  • Multivariate Analysis
  • Neoplasms, Second Primary / chemically induced*
  • Neoplasms, Second Primary / epidemiology
  • Neoplasms, Second Primary / pathology
  • Nitriles
  • Primary Myelofibrosis / drug therapy*
  • Primary Myelofibrosis / pathology
  • Pyrazoles / administration & dosage
  • Pyrazoles / adverse effects*
  • Pyrazoles / therapeutic use
  • Pyrazoles / toxicity
  • Pyrimidines
  • Retrospective Studies
  • Risk Factors
  • Skin Neoplasms / diagnosis
  • Skin Neoplasms / epidemiology
  • Thrombocytosis / chemically induced
  • Thrombocytosis / diagnosis
  • Thrombosis / chemically induced
  • Thrombosis / diagnosis

Substances

  • Janus Kinase Inhibitors
  • Nitriles
  • Pyrazoles
  • Pyrimidines
  • ruxolitinib