TY - JOUR
T1 - Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
AU - Kluin-Nelemans, Hanneke C.
AU - Jawhar, Mohamad
AU - Reiter, Andreas
AU - van Anrooij, Bjorn
AU - Gotlib, Jason
AU - Hartmann, Karin
AU - Illerhaus, Anja
AU - Oude Elberink, Hanneke N.G.
AU - Gorska, Aleksandra
AU - Niedoszytko, Marek
AU - Lange, Magdalena
AU - Scaffidi, Luigi
AU - Zanotti, Roberta
AU - Bonadonna, Patrizia
AU - Perkins, Cecelia
AU - Elena, Chiara
AU - Malcovati, Luca
AU - Shoumariyeh, Khalid
AU - von Bubnoff, Nikolas
AU - Müller, Sabine
AU - Triggiani, Massimo
AU - Parente, Roberta
AU - Schwaab, Juliana
AU - Kundi, Michael
AU - Fortina, Anna Belloni
AU - Caroppo, Francesca
AU - Brockow, Knut
AU - Zink, Alexander
AU - Fuchs, David
AU - Angelova-Fischer, Irena
AU - Yavuz, Akif Selim
AU - Doubek, Michael
AU - Mattsson, Mattias
AU - Hagglund, Hans
AU - Panse, Jens
AU - Simonowski, Anne
AU - Sabato, Vito
AU - Schug, Tanja
AU - Jentzsch, Madlen
AU - Breynaert, Christine
AU - Várkonyi, Judit
AU - Kennedy, Vanessa
AU - Hermine, Olivier
AU - Rossignol, Julien
AU - Arock, Michel
AU - Valent, Peter
AU - Sperr, Wolfgang R.
N1 - Funding Information:
This work was supported by the Austrian Science Fund (FWF), SFB grants F4701-B28, F4704-B28, and P32470-B (to P.V.), by the ‘Charles and Ann Johnson Foundation’ (to J.G.). V.S. is a Senior Clinical Researcher of the Research Foundation Flanders/Fonds Wetenschappelijk Onderzoek (FWO: 1804518N). C.B. is supported by the Clinical Research Fund of the University Hospital Leuven.
Funding Information:
Hanneke C. Kluin-Nelemans: institutional financial support from Novartis; honoraria from Novartis for participating in e-learning program. Andreas Reiter: Novartis Pharma – research support, advisory board, honoraria, travel reimbursement, Blueprint Medicines - advisory board, honoraria, travel reimbursement, Deciphera – advisory board, travel reimbursement. Bjorn van Anrooij: financial support from Novartis for research and advisory boards. Jason Gotlib: Funding to support conduct of clinical trial: Blueprint Medicines, Deciphera; advisory board/honoraria: Blueprint Medicines, Deciphera, Allakos. Karin Hartmann: advisory board/honoraria: Allergopharma, ALK-Abelló, Blueprint, Deciphera, Menarini, Novartis and Takeda; research grant: Euroimmun. Hanneke Oude Elberink: honoraria from ALK-Abelló, Chiesi, MEDA Pharma, Novartis, and Blueprint. Magdalena Lange: honoraria from Novartis for lectures. Chiara Elena: advisory board Novartis, Pfizer. David Fuchs, Julien Rossignol: advisory board Novartis. Khalid Shoumariyeh: travel support from Abbvie and consultancy fees from Novartis. Nikolas von Bubnoff: institutional financial support from Novartis. Massimo Triggiani: advisory board BluePrint Medicines, Deciphera, Novartis. Akif SelimYavuz: honoraria from Novartis. Jens Panse: funding to support conduct of clinical trial: Blueprint Medicines, Deciphera; advisory board/honoraria: Blueprint Medicines, Novartis. Vito Sabato: advisory board/honoraria: Blueprint Medicines, Novartis. Madlen Jentzsch: honoraria from Novartis. Olivier Hermine: Research funding support from AB science and Novartis. Advisory board of AB science. Wolfgang R. Sperr: honoraria from Novartis, Pfizer, AbbVie, Daiichi Sankyo, Amgen, Thermo Fisher, Deciphera, Incyte, Celgene and Jazz. Michel Arock: advisory board/honoraria Blueprint Medicines, Deciphera. All other authors: none.
Publisher Copyright:
© The author(s).
Copyright:
Copyright 2020 Elsevier B.V., All rights reserved.
PY - 2020
Y1 - 2020
N2 - In systemic mastocytosis (SM), the clinical features and survival vary greatly. Patient-related factors determining the outcome in SM are largely unknown. Methods: We examined the impact of sex on the clinical features, progression-free survival (PFS), and overall survival (OS) in 3403 patients with mastocytosis collected in the registry of the European Competence Network on Mastocytosis (ECNM). The impact of cytogenetic and molecular genetic aberrations on sex differences was analyzed in a subset of patients. Results: Of all patients enrolled, 55.3% were females. However, a male predominance was found in a subset of advanced SM (AdvSM) patients, namely SM with an associated hematologic neoplasm (SM-AHN, 70%; p < 0.001). Correspondingly, organomegaly (male: 23% vs. female: 13%, p = 0.007) was more, whereas skin involvement (male: 71% vs. female: 86%, p = 0.001) was less frequent in males. In all patients together, OS (p < 0.0001) was significantly inferior in males, and also within the WHO sub-categories indolent SM, aggressive SM (ASM) and SM-AHN. PFS was significantly (p = 0.0002) worse in males when all patients were grouped together; due to low numbers of events, this significance persisted only in the subcategory smoldering SM. Finally, prognostically relevant cytogenetic abnormalities (10% vs. 5%, p = 0.006) or molecular aberrations (SRSF2/ASXL1/RUNX1 profile; 63% vs. 40%, p = 0.003) were more frequently present in males. Conclusions: Male sex has a major impact on clinical features, disease progression, and survival in mastocytosis. Male patients have an inferior survival, which seems related to the fact that they more frequently develop a multi-mutated AdvSM associated with a high-risk molecular background.
AB - In systemic mastocytosis (SM), the clinical features and survival vary greatly. Patient-related factors determining the outcome in SM are largely unknown. Methods: We examined the impact of sex on the clinical features, progression-free survival (PFS), and overall survival (OS) in 3403 patients with mastocytosis collected in the registry of the European Competence Network on Mastocytosis (ECNM). The impact of cytogenetic and molecular genetic aberrations on sex differences was analyzed in a subset of patients. Results: Of all patients enrolled, 55.3% were females. However, a male predominance was found in a subset of advanced SM (AdvSM) patients, namely SM with an associated hematologic neoplasm (SM-AHN, 70%; p < 0.001). Correspondingly, organomegaly (male: 23% vs. female: 13%, p = 0.007) was more, whereas skin involvement (male: 71% vs. female: 86%, p = 0.001) was less frequent in males. In all patients together, OS (p < 0.0001) was significantly inferior in males, and also within the WHO sub-categories indolent SM, aggressive SM (ASM) and SM-AHN. PFS was significantly (p = 0.0002) worse in males when all patients were grouped together; due to low numbers of events, this significance persisted only in the subcategory smoldering SM. Finally, prognostically relevant cytogenetic abnormalities (10% vs. 5%, p = 0.006) or molecular aberrations (SRSF2/ASXL1/RUNX1 profile; 63% vs. 40%, p = 0.003) were more frequently present in males. Conclusions: Male sex has a major impact on clinical features, disease progression, and survival in mastocytosis. Male patients have an inferior survival, which seems related to the fact that they more frequently develop a multi-mutated AdvSM associated with a high-risk molecular background.
UR - http://www.scopus.com/inward/record.url?scp=85094590768&partnerID=8YFLogxK
U2 - 10.7150/THNO.51872
DO - 10.7150/THNO.51872
M3 - Journal articles
C2 - 33391475
AN - SCOPUS:85094590768
SN - 1838-7640
VL - 11
SP - 292
EP - 303
JO - Theranostics
JF - Theranostics
IS - 1
ER -