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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...
Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Ivyspring International Publisher
2021
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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. |
format | Online Article Text |
id | pubmed-7681091 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Ivyspring International Publisher |
record_format | MEDLINE/PubMed |
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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