Prognosis in adult indolent systemic mastocytosis: a long-term study of the Spanish Network on Mastocytosis in a series of 145 patients

J Allergy Clin Immunol. 2009 Sep;124(3):514-21. doi: 10.1016/j.jaci.2009.05.003. Epub 2009 Jul 9.

Abstract

Background: Indolent systemic mastocytosis is a group of rare diseases for which reliable predictors of progression and outcome are still lacking.

Objective: Here we investigate the prognostic impact of the clinical, biological, phenotypic, histopathological, and molecular disease characteristics in adults with indolent systemic mastocytosis, who were followed using conservative therapy.

Methods: A total of 145 consecutive patients were prospectively followed between January 1983 and July 2008; in addition, from 1967 to 1983, 20 patients were retrospectively studied.

Results: Multivariate analysis showed that serum beta2-microglobulin (P = .003) together with the presence of mast/stem cell growth factor receptor gene (KIT) mutation in mast cells plus myeloid and lymphoid hematopoietic lineages (P = .02) was the best combination of independent parameters for predicting disease progression (cumulative probability of disease progression of 1.7% +/- 1.2% at 5-10 years and of 8.4% +/- 5.0% at 20-25 years). Regarding overall survival, the best predictive model included age >60 years (P = .005) and development of an associated clonal hematological non-mast cell disorder (P = .03) with a cumulative probability of death of 2.2% +/- 1.3% at 5 years and of 11% +/- 5.9% at 25 years.

Conclusions: Indolent systemic mastocytosis in adults has a low disease progression rate, and the great majority of patients have a normal life expectancy, with the presence of KIT mutation in all hematopoietic lineages and increased serum beta2-microglobulin the most powerful independent parameters for predicting transformation into a more aggressive form of the disease.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Antiviral Agents / administration & dosage
  • Antiviral Agents / adverse effects
  • Antiviral Agents / therapeutic use
  • Disease Progression
  • Enzyme Inhibitors / administration & dosage
  • Enzyme Inhibitors / adverse effects
  • Enzyme Inhibitors / therapeutic use
  • Female
  • Humans
  • Hydroxyurea / administration & dosage
  • Hydroxyurea / adverse effects
  • Hydroxyurea / therapeutic use
  • Interferon alpha-2
  • Interferon-alpha / administration & dosage
  • Interferon-alpha / adverse effects
  • Interferon-alpha / therapeutic use
  • Male
  • Mast Cells / immunology*
  • Mastocytosis, Systemic / drug therapy
  • Mastocytosis, Systemic / mortality*
  • Mastocytosis, Systemic / pathology*
  • Middle Aged
  • Multivariate Analysis
  • Prognosis
  • Prospective Studies
  • Proto-Oncogene Proteins c-kit / genetics
  • Proto-Oncogene Proteins c-kit / immunology
  • Proto-Oncogene Proteins c-kit / metabolism
  • Recombinant Proteins
  • Retrospective Studies
  • Young Adult
  • beta 2-Microglobulin / blood

Substances

  • Antiviral Agents
  • Enzyme Inhibitors
  • Interferon alpha-2
  • Interferon-alpha
  • Recombinant Proteins
  • beta 2-Microglobulin
  • Proto-Oncogene Proteins c-kit
  • Hydroxyurea