Cytoreductive treatment in patients with CALR-mutated essential thrombocythaemia: a study comparing indications and efficacy among genotypes from the Spanish Registry of Essential Thrombocythaemia

Br J Haematol. 2021 Mar;192(6):988-996. doi: 10.1111/bjh.16988. Epub 2020 Aug 3.

Abstract

The present study assessed the criteria for initiating cytoreduction and response to conventional therapies in 1446 patients with essential thrombocythemia (ET), 267 (17%) of which were CALR-mutated. In low risk patients, time from diagnosis to cytoreduction was shorter in CALR-positive than in the other genotypes (2·8, 3·2, 7·4 and 12·5 years for CALR, MPL, JAK2V617F and TN, respectively, P < 0·0001). A total of 1104 (76%) patients received cytoreductive treatment with hydroxycarbamide (HC) (n = 977), anagrelide (n = 113), or others (n = 14). The estimated cumulative rates of complete haematological response (CR) at 12 months were 40 % and 67% in CALR and JAK2V617F genotypes, respectively. Median time to CR was 192 days for JAK2V617F, 343 for TN, 433 for MPL, and 705 for CALR genotypes (P < 0·0001). Duration of CR was shorter in CALR-mutated ET than in the remaining patients (P = 0·003). In CALR-positive patients, HC and anagrelide had similar efficacy in terms of response rates and duration. CALR-mutated patients developed resistance/intolerance to HC more frequently (5%, 23%, 27% and 15% for JAK2V617F, CALR, MPL and TN, respectively; P < 0·0001). In conclusion, conventional cytoreductive agents are less effective in CALR-mutated ET, highlighting the need for new treatment modalities and redefinition of haematologic targets for patients with this genotype.

Keywords: calreticulin mutation; essential thrombocythaemia; genotype; haematological response; myeloproliferative neoplasms; therapy.

Publication types

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

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Amino Acid Substitution
  • Calreticulin / genetics*
  • Child
  • Female
  • Follow-Up Studies
  • Genotype*
  • Humans
  • Hydroxyurea / administration & dosage*
  • Janus Kinase 2 / genetics
  • Male
  • Middle Aged
  • Mutation, Missense*
  • Quinazolines / administration & dosage*
  • Registries*
  • Spain
  • Thrombocythemia, Essential* / drug therapy
  • Thrombocythemia, Essential* / genetics

Substances

  • CALR protein, human
  • Calreticulin
  • Quinazolines
  • JAK2 protein, human
  • Janus Kinase 2
  • anagrelide
  • Hydroxyurea