Abstract
PURPOSE: To develop a risk score for patients with advanced systemic mastocytosis (AdvSM) that integrates clinical and mutation characteristics. PATIENTS AND METHODS: The study included 383 patients with AdvSM from the German Registry on Disorders of Eosinophils and Mast Cells (training set; n = 231) and several centers for mastocytosis in the United States and Europe, all within the European Competence Network on Mastocytosis (validation set; n = 152). A Cox multivariable model was used to select variables that were predictive of overall survival (OS). RESULTS: In multivariable analysis, the following risk factors were identified as being associated with OS: age greater than 60 years, anemia (hemoglobin, 10 g/dL), thrombocytopenia (platelets, 100 3 109/L), presence of one high molecular risk gene mutation (ie, in SRSF2, ASXL1, and/or RUNX1), and presence of two or more high molecular risk gene mutations. By assigning hazard ratio-weighted points to these variables, the following three risk categories were defined: low risk (median OS, not reached), intermediate risk (median OS, 3.9 years; 95% CI, 2.1 to 5.7 years), and high risk (median OS, 1.9 years; 95% CI, 1.3 to 2.6 years; P, .001). The mutation-adjusted risk score (MARS) was independent of the WHO classification and was confirmed in the independent validation set. During a median follow-up time of 2.2 years (range, 0 to 23 years), 63 (16%) of 383 patients experienced a leukemic transformation to secondary mast cell leukemia (32%) or secondary acute myeloid leukemia (68%). The MARS was also predictive for leukemia-free survival (P, .001). CONCLUSION: The MARS is a validated, five-parameter, WHO-independent prognostic score that defines three risk groups among patients with AdvSM and may improve up-front treatment stratification for these rare hematologic neoplasms.
| Original language | English |
|---|---|
| Journal | Journal of Clinical Oncology |
| Volume | 37 |
| Issue number | 31 |
| Pages (from-to) | 2846-2856 |
| Number of pages | 11 |
| ISSN | 0732-183X |
| DOIs | |
| Publication status | Published - 2019 |
Funding
Supported by the Deutsche Jos? Carreras Leuk?mie-Stiftung (Grant No. 01 R/2018) (M.J.); the SEED program of the Mannheim Medical Faculty, Heidelberg University, Mannheim, Germany (M.J.); Austrian Science Fund Grant No. SFB F4704-B20 (W.R.S. and P.V.); grants from the Instituto de Salud Carlos III and Fondo Europeo de Desarrollo Regional (Grant No. PI16/00642 and Centro de Investigaci?n Biom?dica en Red C?ncer Grant No. CB16/12/00400), Madrid, Spain (I.A.-T., J.I.M.-G., A.C.G.-M., and A.O.); and the Charles and Ann Johnson Foundation (J.G.). Supported by the Deutsche José Carreras Leukämie-Stiftung (Grant No. 01 R/2018) (M.J.); the SEED program of the Mannheim Medical Faculty, Heidelberg University, Mannheim, Germany (M.J.); Austrian Science Fund Grant No. SFB F4704-B20 (W.R.S. and P.V.); grants from the
UN SDGs
This output contributes to the following UN Sustainable Development Goals (SDGs)
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SDG 3 Good Health and Well-being
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