| Literature DB >> 34545185 |
Roberta Zanotti1, Massimiliano Bonifacio2, Giuseppe Lucchini3, Wolfgang R Sperr4,5, Luigi Scaffidi1, Björn van Anrooij6,7, Hanneke Nc Oude Elberink7, Julien Rossignol8, Olivier Hermine8, Aleksandra Gorska9, Magdalena Lange10, Emir Hadzijusufovic4,5,11, Cornelius Miething12, Sabine Müller12, Cecelia Perkins13, William Shomali13, Chiara Elena14, Anja Illerhaus15, Mohamad Jawhar16, Roberta Parente17, Francesca Caroppo18, Oleksii Solomianyi19, Alexander Zink20, Mattias Mattsson21, Akif Selim Yavuz22, Jens Panse23, Judit Varkonyi24, Michael Doubek25, Vito Sabato26, Christine Breynaert27, Vladan Vucinic28, Tanja Schug29, Hans Hägglund21, Friederike Wortmann30, Knut Brockow20, Irena Angelova-Fischer31, Anna Belloni Fortina18, Massimo Triggiani17, Andreas Reiter16, Karin Hartmann32, Luca Malcovati14,33, Jason Gotlib13, Khalid Shoumariyeh12, Marek Niedoszytko9, Michel Arock34, Hanneke C Kluin-Nelemans6, Patrizia Bonadonna35, Peter Valent4,5.
Abstract
In the current classification of the World Health Organization (WHO), bone marrow mastocytosis (BMM) is a provisional variant of indolent systemic mastocytosis (ISM) defined by bone marrow involvement and absence of skin lesions. However, no additional diagnostic criteria for BMM have been proposed. Within the registry dataset of the European Competence Network on Mastocytosis, we compared characteristics and outcomes of 390 patients with BMM and 1175 patients with typical ISM. BMM patients were significantly older, predominantly male, had lower tryptase and lower burden of neoplastic mast cells, and displayed a higher frequency of allergic reactions, mainly triggered by Hymenoptera, than patients with typical ISM. The estimated 10-year progression-free survival of BMM and typical ISM was 95.9% and 92.6%, respectively. In BMM patients defined by WHO-based criteria, the presence of one B-Finding and tryptase level ≥125 ng/mL were identified as risk factors for progression in multivariate analyses. BMM patients without any of these risk factors were found to have better progression-free survival (p < 0.05) and better overall survival (p < 0.05) than other ISM patients. These data support the proposal to define BMM as a separate SM variant characterized by SM criteria, absence of skin lesions, absence of B-Findings, and tryptase levels <125 ng/mL.Entities:
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Year: 2021 PMID: 34545185 DOI: 10.1038/s41375-021-01406-y
Source DB: PubMed Journal: Leukemia ISSN: 0887-6924 Impact factor: 11.528