TY - JOUR
T1 - Mars: Mutation-adjusted risk score for advanced systemic mastocytosis
AU - Jawhar, Mohamad
AU - Schwaab, Juliana
AU - Álvarez-Twose, Iván
AU - Shoumariyeh, Khalid
AU - Naumann, Nicole
AU - Lübke, Johannes
AU - Perkins, Cecelia
AU - Muñoz-González, Javier I.
AU - Meggendorfer, Manja
AU - Kennedy, Vanessa
AU - Metzgeroth, Georgia
AU - Fabarius, Alice
AU - Pfeifer, Dietmar
AU - Sotlar, Karl
AU - Horny, Hans Peter
AU - Von Bubnoff, Nikolas
AU - Haferlach, Torsten
AU - Cross, Nicholas C.P.
AU - Hofmann, Wolf Karsten
AU - Sperr, Wolfgang R.
AU - García-Montero, Andrés C.
AU - Valent, Peter
AU - Gotlib, Jason
AU - Orfao, Alberto
AU - Reiter, Andreas
N1 - Publisher Copyright:
© 2019 by American Society of Clinical Oncology
PY - 2019
Y1 - 2019
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=85074303836&partnerID=8YFLogxK
U2 - 10.1200/JCO.19.00640
DO - 10.1200/JCO.19.00640
M3 - Journal articles
C2 - 31509472
AN - SCOPUS:85074303836
SN - 0732-183X
VL - 37
SP - 2846
EP - 2856
JO - Journal of Clinical Oncology
JF - Journal of Clinical Oncology
IS - 31
ER -