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Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis

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...

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Autores principales: Kluin-Nelemans, Hanneke C., Jawhar, Mohamad, Reiter, Andreas, van Anrooij, Bjorn, Gotlib, Jason, Hartmann, Karin, Illerhaus, Anja, Oude Elberink, Hanneke N.G., Gorska, Aleksandra, Niedoszytko, Marek, Lange, Magdalena, Scaffidi, Luigi, Zanotti, Roberta, Bonadonna, Patrizia, Perkins, Cecelia, Elena, Chiara, Malcovati, Luca, Shoumariyeh, Khalid, von Bubnoff, Nikolas, Müller, Sabine, Triggiani, Massimo, Parente, Roberta, Schwaab, Juliana, Kundi, Michael, Fortina, Anna Belloni, Caroppo, Francesca, Brockow, Knut, Zink, Alexander, Fuchs, David, Angelova-Fischer, Irena, Yavuz, Akif Selim, Doubek, Michael, Mattsson, Mattias, Hagglund, Hans, Panse, Jens, Simonowski, Anne, Sabato, Vito, Schug, Tanja, Jentzsch, Madlen, Breynaert, Christine, Várkonyi, Judit, Kennedy, Vanessa, Hermine, Olivier, Rossignol, Julien, Arock, Michel, Valent, Peter, Sperr, Wolfgang R.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Ivyspring International Publisher 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7681091/
https://www.ncbi.nlm.nih.gov/pubmed/33391475
http://dx.doi.org/10.7150/thno.51872
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author Kluin-Nelemans, Hanneke C.
Jawhar, Mohamad
Reiter, Andreas
van Anrooij, Bjorn
Gotlib, Jason
Hartmann, Karin
Illerhaus, Anja
Oude Elberink, Hanneke N.G.
Gorska, Aleksandra
Niedoszytko, Marek
Lange, Magdalena
Scaffidi, Luigi
Zanotti, Roberta
Bonadonna, Patrizia
Perkins, Cecelia
Elena, Chiara
Malcovati, Luca
Shoumariyeh, Khalid
von Bubnoff, Nikolas
Müller, Sabine
Triggiani, Massimo
Parente, Roberta
Schwaab, Juliana
Kundi, Michael
Fortina, Anna Belloni
Caroppo, Francesca
Brockow, Knut
Zink, Alexander
Fuchs, David
Angelova-Fischer, Irena
Yavuz, Akif Selim
Doubek, Michael
Mattsson, Mattias
Hagglund, Hans
Panse, Jens
Simonowski, Anne
Sabato, Vito
Schug, Tanja
Jentzsch, Madlen
Breynaert, Christine
Várkonyi, Judit
Kennedy, Vanessa
Hermine, Olivier
Rossignol, Julien
Arock, Michel
Valent, Peter
Sperr, Wolfgang R.
author_facet Kluin-Nelemans, Hanneke C.
Jawhar, Mohamad
Reiter, Andreas
van Anrooij, Bjorn
Gotlib, Jason
Hartmann, Karin
Illerhaus, Anja
Oude Elberink, Hanneke N.G.
Gorska, Aleksandra
Niedoszytko, Marek
Lange, Magdalena
Scaffidi, Luigi
Zanotti, Roberta
Bonadonna, Patrizia
Perkins, Cecelia
Elena, Chiara
Malcovati, Luca
Shoumariyeh, Khalid
von Bubnoff, Nikolas
Müller, Sabine
Triggiani, Massimo
Parente, Roberta
Schwaab, Juliana
Kundi, Michael
Fortina, Anna Belloni
Caroppo, Francesca
Brockow, Knut
Zink, Alexander
Fuchs, David
Angelova-Fischer, Irena
Yavuz, Akif Selim
Doubek, Michael
Mattsson, Mattias
Hagglund, Hans
Panse, Jens
Simonowski, Anne
Sabato, Vito
Schug, Tanja
Jentzsch, Madlen
Breynaert, Christine
Várkonyi, Judit
Kennedy, Vanessa
Hermine, Olivier
Rossignol, Julien
Arock, Michel
Valent, Peter
Sperr, Wolfgang R.
author_sort Kluin-Nelemans, Hanneke C.
collection PubMed
description 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.
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spelling pubmed-76810912021-01-01 Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis Kluin-Nelemans, Hanneke C. Jawhar, Mohamad Reiter, Andreas van Anrooij, Bjorn Gotlib, Jason Hartmann, Karin Illerhaus, Anja Oude Elberink, Hanneke N.G. Gorska, Aleksandra Niedoszytko, Marek Lange, Magdalena Scaffidi, Luigi Zanotti, Roberta Bonadonna, Patrizia Perkins, Cecelia Elena, Chiara Malcovati, Luca Shoumariyeh, Khalid von Bubnoff, Nikolas Müller, Sabine Triggiani, Massimo Parente, Roberta Schwaab, Juliana Kundi, Michael Fortina, Anna Belloni Caroppo, Francesca Brockow, Knut Zink, Alexander Fuchs, David Angelova-Fischer, Irena Yavuz, Akif Selim Doubek, Michael Mattsson, Mattias Hagglund, Hans Panse, Jens Simonowski, Anne Sabato, Vito Schug, Tanja Jentzsch, Madlen Breynaert, Christine Várkonyi, Judit Kennedy, Vanessa Hermine, Olivier Rossignol, Julien Arock, Michel Valent, Peter Sperr, Wolfgang R. Theranostics Research Paper 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. Ivyspring International Publisher 2021-01-01 /pmc/articles/PMC7681091/ /pubmed/33391475 http://dx.doi.org/10.7150/thno.51872 Text en © The author(s) This is an open access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/). See http://ivyspring.com/terms for full terms and conditions.
spellingShingle Research Paper
Kluin-Nelemans, Hanneke C.
Jawhar, Mohamad
Reiter, Andreas
van Anrooij, Bjorn
Gotlib, Jason
Hartmann, Karin
Illerhaus, Anja
Oude Elberink, Hanneke N.G.
Gorska, Aleksandra
Niedoszytko, Marek
Lange, Magdalena
Scaffidi, Luigi
Zanotti, Roberta
Bonadonna, Patrizia
Perkins, Cecelia
Elena, Chiara
Malcovati, Luca
Shoumariyeh, Khalid
von Bubnoff, Nikolas
Müller, Sabine
Triggiani, Massimo
Parente, Roberta
Schwaab, Juliana
Kundi, Michael
Fortina, Anna Belloni
Caroppo, Francesca
Brockow, Knut
Zink, Alexander
Fuchs, David
Angelova-Fischer, Irena
Yavuz, Akif Selim
Doubek, Michael
Mattsson, Mattias
Hagglund, Hans
Panse, Jens
Simonowski, Anne
Sabato, Vito
Schug, Tanja
Jentzsch, Madlen
Breynaert, Christine
Várkonyi, Judit
Kennedy, Vanessa
Hermine, Olivier
Rossignol, Julien
Arock, Michel
Valent, Peter
Sperr, Wolfgang R.
Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title_full Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title_fullStr Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title_full_unstemmed Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title_short Cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
title_sort cytogenetic and molecular aberrations and worse outcome for male patients in systemic mastocytosis
topic Research Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7681091/
https://www.ncbi.nlm.nih.gov/pubmed/33391475
http://dx.doi.org/10.7150/thno.51872
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