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| Título: | Nonaggressive systemic mastocytosis (SM) without skin lesions associated with insect-induced anaphylaxis shows unique features versus other indolent SM |
Autor: | Álvarez-Twose, Iván; Zanotti, Roberta; Gonzalez-de-Olano, David; Matito, Almudena; Sánchez-Muñoz, Laura; Morgado, J. M.; García-Montero, Andrés CSIC ORCID; Teodosio, Cristina; Jara-Acevedo, Maria; Mayado, Andrea; Mollejo, Manuela; Orfao, Alberto CSIC ORCID ; Escribano, Luis | Fecha de publicación: | 2014 | Editor: | Elsevier | Citación: | Journal of Allergy and Clinical Immunology 133(2): 520-528.e5 (2014) | Resumen: | [Background]: Indolent systemic mastocytosis (ISM) without skin lesions (ISMs-) shows a higher prevalence in males, lower serum baseline tryptase levels, and KIT mutation more frequently restricted to bone marrow (BM) mast cells (MCs) than ISM with skin lesions (ISMs+). Interestingly, in almost one-half of ISMs- patients, MC-mediator release episodes are triggered exclusively by insects. [Objective]: We aimed to determine the clinical and laboratory features of ISMs- associated with insect-induced anaphylaxis (insectISMs-) versus other patients with ISM. [Methods]: A total of 335 patients presenting with MC activation syndrome, including 143 insectISMs-, 72 ISMs- triggered by other factors (otherISMs-), 56 ISMs+, and 64 nonclonal MC activation syndrome, were studied. [Results]: Compared with otherISMs- and ISMs+ patients, insectISMs- cases showed marked male predominance (78% vs 53% and 46%; P <.001), a distinct pattern of MC-related symptoms, and significantly lower median serum baseline tryptase levels (22.4 vs 28.7 and 45.8 μg/L; P ≤.009). Moreover, insectISMs- less frequently presented BM MC aggregates (46% vs 70% and 81%; P ≤.001), and they systematically showed MC-restricted KIT mutation. [Conclusions]: ISMs- patients with anaphylaxis triggered exclusively by insects display clinical and laboratory features that are significantly different from other ISM cases, including other ISMs- and ISMs+ patients, suggesting that they represent a unique subgroup of ISM with a particularly low BM MC burden in the absence of adverse prognostic factors. | Descripción: | Spanish Network on Mastocytosis (REMA): et al. | URI: | http://hdl.handle.net/10261/134753 | DOI: | 10.1016/j.jaci.2013.06.020 | Identificadores: | doi: 10.1016/j.jaci.2013.06.020 issn: 0091-6749 e-issn: 1097-6825 |
| Aparece en las colecciones: | (IBMCC) Artículos |
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