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51. Small airways function: evaluation in a population of adult patients with severe asthma and potential use as a response biomarker for anti-IL5 therapy

Author

Maglio, Angelantonio, primary, Vitale, Carolina, additional, Pellegrino, Simona, additional, Calabrese, Cecilia, additional, Parente, Roberta, additional, Triggiani, Massimo, additional, Stellato, Cristiana, additional, Pelaia, Corrado, additional, Pelaia, Girolamo, additional, and Vatrella, Alessandro, additional

Published
2020
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52. Clinical features and survival of patients with indolent systemic mastocytosis defined by the updated WHO classification

Author

Trizuljak, Jakub, primary, Sperr, Wolfgang R., additional, Nekvindová, Lucie, additional, Elberink, Hanneke O., additional, Gleixner, Karoline V., additional, Gorska, Aleksandra, additional, Lange, Magdalena, additional, Hartmann, Karin, additional, Illerhaus, Anja, additional, Bonifacio, Massimiliano, additional, Perkins, Cecelia, additional, Elena, Chiara, additional, Malcovati, Luca, additional, Fortina, Anna B., additional, Shoumariyeh, Khalid, additional, Jawhar, Mohamad, additional, Zanotti, Roberta, additional, Bonadonna, Patrizia, additional, Caroppo, Francesca, additional, Zink, Alexander, additional, Triggiani, Massimo, additional, Parente, Roberta, additional, von Bubnoff, Nikolas, additional, Yavuz, Akif S., additional, Hägglund, Hans, additional, Mattsson, Mattias, additional, Panse, Jens, additional, Jäkel, Nadja, additional, Kilbertus, Alex, additional, Hermine, Olivier, additional, Arock, Michel, additional, Fuchs, David, additional, Sabato, Vito, additional, Brockow, Knut, additional, Bretterklieber, Agnes, additional, Niedoszytko, Marek, additional, van Anrooij, Björn, additional, Reiter, Andreas, additional, Gotlib, Jason, additional, Kluin‐Nelemans, Hanneke C., additional, Mayer, Jiri, additional, Doubek, Michael, additional, and Valent, Peter, additional

Published
2020
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53. Prognostic impact of eosinophils in mastocytosis: analysis of 2350 patients collected in the ECNM Registry

Author

Kluin-Nelemans, Hanneke C., primary, Reiter, Andreas, additional, Illerhaus, Anja, additional, van Anrooij, Bjorn, additional, Hartmann, Karin, additional, Span, Lambertus F. R., additional, Gorska, Aleksandra, additional, Niedoszytko, Marek, additional, Lange, Magdalena, additional, Scaffidi, Luigi, additional, Zanotti, Roberta, additional, Bonadonna, Patrizia, additional, Perkins, Cecelia, additional, Elena, Chiara, additional, Malcovati, Luca, additional, Shoumariyeh, Khalid, additional, von Bubnoff, Nikolas, additional, Parente, Roberta, additional, Triggiani, Massimo, additional, Schwaab, Juliana, additional, Jawhar, Mohamad, additional, Caroppo, Francesca, additional, Fortina, Anna Belloni, additional, Brockow, Knut, additional, Zink, Alexander, additional, Fuchs, David, additional, Kilbertus, Alex, additional, Yavuz, Akif Selim, additional, Doubek, Michael, additional, Mattsson, Mattias, additional, Hagglund, Hans, additional, Panse, Jens, additional, Sabato, Vito, additional, Aberer, Elisabeth, additional, Niederwieser, Dietger, additional, Breynaert, Christine, additional, Várkonyi, Judit, additional, Kennedy, Vanessa, additional, Lortholary, Olivier, additional, Jakob, Thilo, additional, Hermine, Olivier, additional, Rossignol, Julien, additional, Arock, Michel, additional, Gotlib, Jason, additional, Valent, Peter, additional, and Sperr, Wolfgang R., additional

Published
2019
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54. International prognostic scoring system for mastocytosis (IPSM) : a retrospective cohort study

Author

Sperr, Wolfgang R., Kundi, Michael, Alvarez-Twose, Ivan, van Anrooij, Bjorn, Elberink, Joanna N. G. Oude, Gorska, Aleksandra, Niedoszytko, Marek, Gleixner, Karoline V., Hadzijusufovic, Emir, Zanotti, Roberta, Bonadonna, Patrizia, Bonifacio, Massimiliano, Perkins, Cecelia, Illerhaus, Anja, Elena, Chiara, Merante, Serena, Shoumariyeh, Khalid, von Bubnoff, Nikolas, Parente, Roberta, Jawhar, Mohamad, Fortina, Anna Belloni, Caroppo, Francesca, Brockow, Knut, Zink, Alexander, Fuchs, David, Kilbertus, Alex J., Yavuz, Akif Selim, Doubek, Michael, Hägglund, Hans, Panse, Jens, Sabato, Vito, Bretterklieber, Agnes, Niederwieser, Dietger, Breynaert, Christine, Hartmann, Karin, Triggiani, Massimo, Nedoszytko, Boguslaw, Reiter, Andreas, Orfao, Alberto, Hermine, Olivier, Gotlib, Jason, Arock, Michel, Kluin-Nelemans, Hanneke C., Valent, Peter, Sperr, Wolfgang R., Kundi, Michael, Alvarez-Twose, Ivan, van Anrooij, Bjorn, Elberink, Joanna N. G. Oude, Gorska, Aleksandra, Niedoszytko, Marek, Gleixner, Karoline V., Hadzijusufovic, Emir, Zanotti, Roberta, Bonadonna, Patrizia, Bonifacio, Massimiliano, Perkins, Cecelia, Illerhaus, Anja, Elena, Chiara, Merante, Serena, Shoumariyeh, Khalid, von Bubnoff, Nikolas, Parente, Roberta, Jawhar, Mohamad, Fortina, Anna Belloni, Caroppo, Francesca, Brockow, Knut, Zink, Alexander, Fuchs, David, Kilbertus, Alex J., Yavuz, Akif Selim, Doubek, Michael, Hägglund, Hans, Panse, Jens, Sabato, Vito, Bretterklieber, Agnes, Niederwieser, Dietger, Breynaert, Christine, Hartmann, Karin, Triggiani, Massimo, Nedoszytko, Boguslaw, Reiter, Andreas, Orfao, Alberto, Hermine, Olivier, Gotlib, Jason, Arock, Michel, Kluin-Nelemans, Hanneke C., and Valent, Peter

Abstract

Background The WHO classification separates mastocytosis into distinct variants, but prognostication remains a clinical challenge. The aim of this study was to improve prognostication for patients with mastocytosis. Methods We analysed data of the registry of the European Competence Network on Mastocytosis including 1639 patients (age 17-90 years) diagnosed with mastocytosis according to WHO criteria between Jan 12, 1978, and March 16, 2017. Univariate and multivariate analyses with Cox regression were applied to identify prognostic variables predicting survival outcomes and to establish a prognostic score. We validated this International Prognostic Scoring System in Mastocytosis (IPSM) with data of 462 patients (age 17-79 years) from the Spanish network Red Espanola de Mastocitosis diagnosed between Jan 22, 1998, and Nov 2, 2017. Findings The prognostic value of the WHO classification was confirmed in our study (p<0.0001). For patients with non-advanced mastocytosis (n=1380), we identified age 60 years or older (HR 10.75, 95% CI 5.68-20.32) and a concentration of alkaline phosphatase 100 U/L or higher (2.91, 1.60-5.30) as additional independent prognostic variables for overall survival. The resulting scoring system divided patients with non-advanced mastocytosis into three groups: low (no risk factors), intermediate 1 (one risk factor), and intermediate 2 (two risk factors). Overall survival and progression-free survival differed significantly among these groups (p<0.0001). In patients with advanced mastocytosis (n=259), age 60 years or older (HR 2.14, 95% CI 1.42-3.22), a concentration of tryptase 125 ng/mL or higher (1.81, 1.20-2.75), a leukocyte count of 16 x 10(9) per L or higher (1.88, 1.27-2.79), haemoglobin of 11 g/dL or lower (1.71, 1.13-2.57), a platelet count of 100 x 10(9) per L or lower (1.63, 1.13-2.34), and skin involvement (0.46, 0.30-0.69) were prognostic variables. Based on these variables, a separate score for advanced mastocytosis with four

Published
2019
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55. International prognostic scoring system for mastocytosis (IPSM): a retrospective cohort study

Author

Austrian Science Fund, German Research Foundation, University of Cologne, Charles and Ann Johnson Foundation, Research Foundation - Flanders, University of Leuven, Instituto de Salud Carlos III, European Commission, Sperr, Wolfgang R., Kundi, Michael, Anrooij, Bjorn van, Oude Elberink, Joanna N. G., Gorska, Aleksandra, Niedoszytko, Marek, Gleixner, Karoline V., Hadzijusufovic, Emir, Zanotti, Roberta, Bonadonna, Patrizia, Bonifacio, Massimiliano, Perkins, Cecelia, Illerhaus, Anja, Elena, Chiara, Merante, Serena, Shoumariyeh, Khalid, Bubnoff, Nikolas von, Parente, Roberta, Jawhar, Mohamad, Belloni Fortina, Anna, Caroppo, Francesca, Brockow, Knut, Zink, Alexander, Fuchs, David, Kilbertus, Alex J., Selim Yavuz, Akif, Doubek, Michael, Hägglund, Hans, Panse, Jens, Sabato, Vito, Bretterklieber, Agnes, Niederwieser, Dietger, Breynaert, Christine, Hartmann, Karin, Triggiani, Massimo, Nedoszytko, Boguslaw, Reiter, Andreas, Orfao, Alberto, Hermine, Olivier, Gotlib, Jason, Arock, Michel, Kluin-Nelemans, Hanneke C., Valent, Peter, Austrian Science Fund, German Research Foundation, University of Cologne, Charles and Ann Johnson Foundation, Research Foundation - Flanders, University of Leuven, Instituto de Salud Carlos III, European Commission, Sperr, Wolfgang R., Kundi, Michael, Anrooij, Bjorn van, Oude Elberink, Joanna N. G., Gorska, Aleksandra, Niedoszytko, Marek, Gleixner, Karoline V., Hadzijusufovic, Emir, Zanotti, Roberta, Bonadonna, Patrizia, Bonifacio, Massimiliano, Perkins, Cecelia, Illerhaus, Anja, Elena, Chiara, Merante, Serena, Shoumariyeh, Khalid, Bubnoff, Nikolas von, Parente, Roberta, Jawhar, Mohamad, Belloni Fortina, Anna, Caroppo, Francesca, Brockow, Knut, Zink, Alexander, Fuchs, David, Kilbertus, Alex J., Selim Yavuz, Akif, Doubek, Michael, Hägglund, Hans, Panse, Jens, Sabato, Vito, Bretterklieber, Agnes, Niederwieser, Dietger, Breynaert, Christine, Hartmann, Karin, Triggiani, Massimo, Nedoszytko, Boguslaw, Reiter, Andreas, Orfao, Alberto, Hermine, Olivier, Gotlib, Jason, Arock, Michel, Kluin-Nelemans, Hanneke C., and Valent, Peter

Abstract

[Background]: The WHO classification separates mastocytosis into distinct variants, but prognostication remains a clinical challenge. The aim of this study was to improve prognostication for patients with mastocytosis., [Methods]: We analysed data of the registry of the European Competence Network on Mastocytosis including 1639 patients (age 17–90 years) diagnosed with mastocytosis according to WHO criteria between Jan 12, 1978, and March 16, 2017. Univariate and multivariate analyses with Cox regression were applied to identify prognostic variables predicting survival outcomes and to establish a prognostic score. We validated this International Prognostic Scoring System in Mastocytosis (IPSM) with data of 462 patients (age 17–79 years) from the Spanish network Red Española de Mastocitosis diagnosed between Jan 22, 1998, and Nov 2, 2017., [Findings]: The prognostic value of the WHO classification was confirmed in our study (p<0·0001). For patients with non-advanced mastocytosis (n=1380), we identified age 60 years or older (HR 10·75, 95% CI 5·68–20·32) and a concentration of alkaline phosphatase 100 U/L or higher (2·91, 1·60–5·30) as additional independent prognostic variables for overall survival. The resulting scoring system divided patients with non-advanced mastocytosis into three groups: low (no risk factors), intermediate 1 (one risk factor), and intermediate 2 (two risk factors). Overall survival and progression-free survival differed significantly among these groups (p<0·0001). In patients with advanced mastocytosis (n=259), age 60 years or older (HR 2·14, 95% CI 1·42–3·22), a concentration of tryptase 125 ng/mL or higher (1·81, 1·20–2·75), a leukocyte count of 16 × 109 per L or higher (1·88, 1·27–2·79), haemoglobin of 11 g/dL or lower (1·71, 1·13–2·57), a platelet count of 100 × 109 per L or lower (1·63, 1·13–2·34), and skin involvement (0·46, 0·30–0·69) were prognostic variables. Based on these variables, a separate score for advanced mastocytosis with four risk categories was established, with significantly different outcomes for overall survival and progression-free survival (p<0·0001). The prognostic value of both scores was confirmed in 413 patients with non-advanced disease and 49 with advanced mastocytosis from the validation cohort., [Interpretation]: The IPSM scores for patients with non-advanced and advanced mastocytosis can be used to predict survival outcomes and guide treatment decisions. However, the predictive value of the IPSM needs to be confirmed in forthcoming trials.

Published
2019

56. Pore-forming peptides induce rapid phospholipid flip-flop in membranes

Author

Fattal, Elias, Nir, Shlomo, Parente, Roberta A., and Szoka, Francis C., Jr.

Subjects
Peptides -- Research, Phospholipids -- Physiological aspects, Biological sciences, Chemistry
Abstract

Amphipathic segments arranging into transmembrane oligomeric pores regulate non-energy-dependent phospholipid flip-flop in biological membranes. Certain peptides group within a membrane and reach a critical size when a channel formed results in a fast flip-flop of phospholipids. Membrane-relating peptides such as LAGA, unable to form pores, accelerate flip-flop through bilayer disturbances.

Published
1994

57. Altered Metabolism of Phospholipases, Diacylglycerols, Endocannabinoids, and N-Acylethanolamines in Patients with Mastocytosis

Author

Ferrara, Anne Lise, primary, Piscitelli, Fabiana, additional, Petraroli, Angelica, additional, Parente, Roberta, additional, Galdiero, Maria Rosaria, additional, Varricchi, Gilda, additional, Marone, Giancarlo, additional, Triggiani, Massimo, additional, Di Marzo, Vincenzo, additional, and Loffredo, Stefania, additional

Published
2019
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58. The Severe Asthma Network in Italy: Findings and Perspectives

Author

Heffler, Enrico, primary, Blasi, Francesco, additional, Latorre, Manuela, additional, Menzella, Francesco, additional, Paggiaro, Pierluigi, additional, Pelaia, Girolamo, additional, Senna, Gianenrico, additional, Canonica, Giorgio Walter, additional, Barbuto, Sarah, additional, Bradicich, Matteo, additional, Camiciottoli, Gianna, additional, Caminati, Marco, additional, Colombo, Giselda, additional, Costantino, Maria Teresa, additional, Crimi, Nunzio, additional, Crivellaro, Mariangiola, additional, D’Adda, Alice, additional, D’Amato, Mariella, additional, Favero, Elisabetta, additional, Foschino, Maria Pia, additional, Galeone, Carla, additional, Guarnieri, Gabriella, additional, Lombardi, Carlo, additional, Parente, Roberta, additional, Passalacqua, Giovanni, additional, Patella, Vincenzo, additional, Puggioni, Francesca, additional, Ridolo, Erminia, additional, Rolla, Giovanni, additional, Savi, Eleonora, additional, Scichilone, Nicola, additional, Solidoro, Paolo, additional, and Spadaro, Giuseppe, additional

Published
2019
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59. Distinct Diagnostic and Clinical Features of Bone Marrow Mastocytosis: A Study of the Registry of the European Competence Network on Mastocytosis (ECNM)

Author

Bonifacio, Massimiliano, Bonadonna, Patrizia, Elberink, Hanneke Oude, Anrooij, Bjorn, Gorska, Aleksandra, Lange, Magdalena, Hadzijusufovic, Emir, Scaffidi, Luigi, Tanasi, Ilaria, Perkins, Cecelia, Virginia Valeria Ferretti, Illerhaus, Anja, Jakob, Thilo, Parente, Roberta, Jawhar, Mohamad, Caroppo, Francesca, Zink, Alexander, Kilbertus, Alex, Yavuz, Akif Selim, Doubek, Michael, Hagglund, Hans, Panse, Jens P., Sabato, Vito, Aberer, Elisabeth, Jaekel, Nadja, Vandenberghe, Peter, Fuchs, David, Brockow, Knut, Fortina, Anna Belloni, Reiter, Andreas, Triggiani, Massimo, Shoumariyeh, Khalid, Hartmann, Karin, Elena, Chiara, Hermine, Olivier, Gotlib, Jason, Niedoszytko, Marek, Kluin-Nelemans, Hanneke, Sperr, Wolfgang R., Valent, Peter, and Zanotti, Roberta

Published
2017

62. Living with chronic spontaneous urticaria in Italy: A narrative medicine project to improve the pathway of patient care

Author

Cappuccio, Antonietta, Limonta, Tommaso, Parodi, Aurora, Cristaudo, Antonio, Bugliaro, Filomena, Cannavò, Serafinella P., Rossi, Oliviero, Gurioli, Carlotta, Vignoli, Alice, Parente, Roberta, Iemoli, Enrico, Caldarola, Giacomo, De Pità, Ornella, Di Nuzzo, Sergio, Cancian, Mauro, Potenza, Concetta, Caminati, Marco, Stingeni, Luca, Saraceno, Rosita, Trevisini, Sara, Piccirillo, Angelo, Sciarrone, Claudio, Panebianco, Rosanna, Gola, Massimo, Costanzo, Antonio, Grieco, Teresa, Massaroni, Katia, Reale, Luigi, Marini, Maria G., Caldarola, Giacomo (ORCID:0000-0002-8837-9232), Cappuccio, Antonietta, Limonta, Tommaso, Parodi, Aurora, Cristaudo, Antonio, Bugliaro, Filomena, Cannavò, Serafinella P., Rossi, Oliviero, Gurioli, Carlotta, Vignoli, Alice, Parente, Roberta, Iemoli, Enrico, Caldarola, Giacomo, De Pità, Ornella, Di Nuzzo, Sergio, Cancian, Mauro, Potenza, Concetta, Caminati, Marco, Stingeni, Luca, Saraceno, Rosita, Trevisini, Sara, Piccirillo, Angelo, Sciarrone, Claudio, Panebianco, Rosanna, Gola, Massimo, Costanzo, Antonio, Grieco, Teresa, Massaroni, Katia, Reale, Luigi, Marini, Maria G., and Caldarola, Giacomo (ORCID:0000-0002-8837-9232)

Abstract

Chronic spontaneous urticaria (CSU) is perceived as a difficult to manage disease with negative impact on quality of life. The aim of this study was to highlight how to improve the care of people with CSU, using the methodology of narrative medicine. From June 2014 to March 2015, CSU-diagnosed patients and their physicians were asked to record their experiences of the condition in writing. Fourteen healthcare teams participated: 41% considered CSU as a challenge to overcome, while 22% experienced CSU as a big commitment. The number of professional involved was evaluated as insufficient in 11 hospitals. Seventy-five percent of the 190 Italian patients had visited 3 or more physicians before receiving a final diagnosis, with a perceived waste of time and resources. The therapeutic pathways were described as unsatisfactory in 83% of cases. As a result, anger and frustration were life-dominant emotions in 92% of patients. The critical points of the care pathway are related to organizational issues and lack of awareness.

Published
2017

66. Altered Metabolism of Phospholipases, Diacylglycerols, Endocannabinoids, and -Acylethanolamines in Patients with Mastocytosis.

Author

Ferrara, Anne Lise, Piscitelli, Fabiana, Petraroli, Angelica, Parente, Roberta, Galdiero, Maria Rosaria, Varricchi, Gilda, Marone, Giancarlo, Triggiani, Massimo, Di Marzo, Vincenzo, and Loffredo, Stefania

Subjects
MAST cell disease, PHOSPHOLIPASES, TRYPTASE, DIGLYCERIDES, MAST cells, METABOLISM
Abstract

Background: Mastocytosis is a condition characterized by the expansion and accumulation of mast cells (MCs) in various organs. The symptoms are related to the increased release of MC-derived mediators that exert local and distant effects. MCs are a source and target of phospholipase enzymes (PLs), which catalyze the cleavage of membrane phospholipids releasing lipid mediators (e.g., diacylglycerols (DAGs) and the endocannabinoid (EC) 2-arachidonoylglycerol (2-AG)). To date, there are no data on the role of these lipid mediators in mastocytosis. Here, we analyzed plasma levels of PLA2, PLC, DAG, ECs, and EC-related N-acylethanolamines in patients with mastocytosis.Methods: In 23 patients with mastocytosis and 23 healthy individuals, we measured plasma PLA2 and PLC activities, DAG, 2-AG, anandamide (AEA), palmitoylethanolamide (PEA), and oleoylethanolamide (OEA).Results: Plasma PLA2 and PLC activities were increased in mastocytosis patients compared to controls. Concentrations of DAG (18:1 20:4 and 18:0 20:4), two second messengers produced by PLC, were higher in mastocytosis compared to controls, whereas the concentrations of their metabolite, 2-AG, were not altered. AEA was decreased in mastocytosis patients compared to controls; by contrast, AEA congener, PEA, was increased. PLA2 and PLC activities were increased only in patients with mediator-related symptoms. Moreover, PLC activity was positively correlated with disease severity and tryptase concentrations. By contrast, AEA was negatively correlated with tryptase concentrations.Conclusions: PLs and some lipid mediators are altered in patients with mastocytosis. Our results may pave the way for investigating the functions of these mediators in the pathophysiology of mastocytosis and provide new potential biomarkers and therapeutic targets. [ABSTRACT FROM AUTHOR]

Published
2019
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68. Selection and Efficacy of Cytoreductive Agents in Patients with Mastocytosis Included in the Registry of the European Competence Network on Mastocytosis (ECNM)

Author

Elena, Chiara, Merante, Serena, Ferretti, Virginia Valeria, Span, Lambertus CM, Górska, Aleksandra, Bonifacio, Massimiliano, Perkins, Cecelia, Hermine, Olivier, Illerhaus, Anja, Mueller, Sabine, Parente, Roberta, Jawhar, Mohamad, Fontana, Elena, Zink, Alexander, Kilbertus, Alex, Yavuz, Akif Selim, Doubek, Michael, Hagglund, Hans, Panse, Jens P., Sabato, Vito, Al Ali, Haifa Kathrin, Aberer, Elisabeth, Morren, Marie Anne, Lange, Magdalena, Fuchs, David, Brockow, Knut, Belloni Fortina, Anna, Reiter, Andreas, Triggiani, Massimo, von Bubnoff, Nikolas, Hartmann, Karin, Gotlib, Jason, Scaffidi, Luigi, Niedoszytko, Marek, Kluin-Nelemans, Hanneke, Malcovati, Luca, Sperr, Wolfgang R., and Valent, Peter

Published
2017
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81. Quality of life in patients with allergic and immunologic skin diseases: in the eye of the beholder

Author

Ester Di Agosta, Lorenzo Salvati, Monica Corazza, Ilaria Baiardini, Francesca Ambrogio, Luisa Angileri, Elettra Antonelli, Federica Belluzzo, Domenico Bonamonte, Laura Bonzano, Raffaele Brancaccio, Paolo Custurone, Aurora De Marco, Aikaterini Detoraki, Adriana Di Guida, Elisabetta Di Leo, Marta Fantò, Filippo Fassio, Silvia Mariel Ferrucci, Caterina Foti, Rosella Gallo, Alessia Gatta, Fabrizio Guarneri, Lucia Guidolin, Katharina Hansel, Donatella Lamacchia, Carla Lombardo, Paola Lucia Minciullo, Maddalena Napolitano, Alessandro Pannofino, Andrea Paravisi, Roberta Parente, Maria Passante, Cataldo Patruno, Diego Peroni, Cristina Quecchia, Natale Schettini, Giuseppe Spadaro, Luca Stingeni, Daniele Tarrini, Marta Tramontana, Eustachio Nettis, Oliviero Rossi, Di Agosta, Ester, Salvati, Lorenzo, Corazza, Monica, Baiardini, Ilaria, Ambrogio, Francesca, Angileri, Luisa, Antonelli, Elettra, Belluzzo, Federica, Bonamonte, Domenico, Bonzano, Laura, Brancaccio, Raffaele, Custurone, Paolo, De Marco, Aurora, Detoraki, Aikaterini, DI GUIDA, Adriana, Di Leo, Elisabetta, Fantò, Marta, Fassio, Filippo, Mariel Ferrucci, Silvia, Foti, Caterina, Gallo, Rosella, Gatta, Alessia, Guarneri, Fabrizio, Guidolin, Lucia, Hansel, Katharina, Lamacchia, Donatella, Lombardo, Carla, Lucia Minciullo, Paola, Napolitano, Maddalena, Pannofino, Alessandro, Paravisi, Andrea, Parente, Roberta, Passante, Maria, Patruno, Cataldo, Peroni, Diego, Quecchia, Cristina, Schettini, Natale, Spadaro, Giuseppe, Stingeni, Luca, Tarrini, Daniele, Tramontana, Marta, Nettis, Eustachio, and Rossi, Oliviero

Subjects
Quality of life, Hereditary angioedema, Cutaneous mastocytosis, Allergy, Urticaria, Allergic contact dermatitis, Atopic dermatitis, Skin, Immunology, Review, RC581-607, humanities, Immunology and Allergy, Immunologic diseases. Allergy, skin and connective tissue diseases, Molecular Biology
Abstract

Allergic and immunologic skin diseases negatively impact the quality of life (QoL) of affected patients with detrimental consequences. Nonetheless, in everyday clinical practice the evaluation of QoL is often overlooked. Considering the increasing prevalence of atopic dermatitis, allergic contact dermatitis, hereditary angioedema, cutaneous mastocytosis, and urticaria, it is essential to determine the effects of allergic and immunologic skin diseases on QoL. A joint meeting (GET TOGETHER 2021) of the Italian Society of Allergology, Asthma and Clinical Immunology (SIAAIC) and the Italian Society of Allergological, Occupational and Environmental Dermatology (SIDAPA) aimed to summarize the features of the main QoL tools used in these diseases and to describe the extent of QoL impairment as well as the impact of treatments on QoL, particularly biologic therapies. The assessment of QoL in patients with allergic and immunologic skin diseases relies on generic, organ-specific and disease-specific questionnaires. While generic and organ-specific questionnaires allow comparison between different diseases, disease-specific questionnaires are designed and validated for specific cohorts: the QoL Index for Atopic Dermatitis (QoLIAD) and the Childhood Atopic Dermatitis Impact Scale (CADIS) in atopic dermatitis, the ACD-11 in allergic contact dermatitis, the Angioedema QoL Questionnaire (AE-QoL) and the Hereditary Angioedema QoL questionnaire (HAE-QoL) in hereditary angioedema, the Mastocytosis QoL Questionnaires (MCQoL e MQLQ) in cutaneous mastocytosis, and the Chronic Urticaria QoL questionnaire (CU-Q2oL) in urticaria. Among the many factors that variably contribute to QoL impairment, pruritus can represent the leading cause of patient discomfort. Biologic therapies significantly ameliorate QoL in atopic dermatitis, hereditary angioedema, mastocytosis and chronic urticaria. In general, adequate management strategies are essential for improving QoL in patients with allergic and immunologic skin diseases.

Published
2021

82. Altered metabolism of phospholipases, diacylglycerols, endocannabinoids, and N-Acylethanolamines in patients with mastocytosis

Author

Anne Lise Ferrara, Giancarlo Marone, Massimo Triggiani, Roberta Parente, Fabiana Piscitelli, Angelica Petraroli, Maria Rosaria Galdiero, Gilda Varricchi, Vincenzo Di Marzo, Stefania Loffredo, Ferrara, Anne Lise, Piscitelli, Fabiana, Petraroli, Angelica, Parente, Roberta, Galdiero, Maria Rosaria, Varricchi, Gilda, Marone, Giancarlo, Triggiani, Massimo, Di Marzo, Vincenzo, and Loffredo, Stefania

Subjects
lcsh:Immunologic diseases. Allergy, Adult, Male, medicine.medical_specialty, Article Subject, Polyunsaturated Alkamides, Immunology, Tryptase, Arachidonic Acids, Phospholipase, Diglycerides, 03 medical and health sciences, chemistry.chemical_compound, Oleoylethanolamide, 0302 clinical medicine, Internal medicine, medicine, Immunology and Allergy, Humans, endocannabinoids, 030304 developmental biology, Aged, 0303 health sciences, Palmitoylethanolamide, biology, Chemistry, General Medicine, Anandamide, Lipid signaling, Middle Aged, Endocannabinoid system, Phospholipases A2, Endocrinology, Ethanolamines, Biomarkers, Endocannabinoids, Female, Mastocytosis, Tryptases, Type C Phospholipases, Second messenger system, biology.protein, lipids (amino acids, peptides, and proteins), lcsh:RC581-607, 030215 immunology
Abstract

Background. Mastocytosis is a condition characterized by the expansion and accumulation of mast cells (MCs) in various organs. The symptoms are related to the increased release of MC-derived mediators that exert local and distant effects. MCs are a source and target of phospholipase enzymes (PLs), which catalyze the cleavage of membrane phospholipids releasing lipid mediators (e.g., diacylglycerols (DAGs) and the endocannabinoid (EC) 2-arachidonoylglycerol (2-AG)). To date, there are no data on the role of these lipid mediators in mastocytosis. Here, we analyzed plasma levels of PLA2, PLC, DAG, ECs, and EC-related N-acylethanolamines in patients with mastocytosis. Methods. In 23 patients with mastocytosis and 23 healthy individuals, we measured plasma PLA2 and PLC activities, DAG, 2-AG, anandamide (AEA), palmitoylethanolamide (PEA), and oleoylethanolamide (OEA). Results. Plasma PLA2 and PLC activities were increased in mastocytosis patients compared to controls. Concentrations of DAG (18:1 20:4 and 18:0 20:4), two second messengers produced by PLC, were higher in mastocytosis compared to controls, whereas the concentrations of their metabolite, 2-AG, were not altered. AEA was decreased in mastocytosis patients compared to controls; by contrast, AEA congener, PEA, was increased. PLA2 and PLC activities were increased only in patients with mediator-related symptoms. Moreover, PLC activity was positively correlated with disease severity and tryptase concentrations. By contrast, AEA was negatively correlated with tryptase concentrations. Conclusions. PLs and some lipid mediators are altered in patients with mastocytosis. Our results may pave the way for investigating the functions of these mediators in the pathophysiology of mastocytosis and provide new potential biomarkers and therapeutic targets.

Published
2019

83. The Severe Asthma Network in Italy: Findings and Perspectives

Author

Giovanni Rolla, Girolamo Pelaia, Mariangiola Crivellaro, Maria Teresa Costantino, Maria Pia Foschino, Enrico Heffler, Alice D'Adda, Carlo Lombardi, Gabriella Guarnieri, Pierluigi Paggiaro, Nunzio Crimi, Francesco Blasi, Eleonora Savi, Manuela Latorre, Carla Galeone, Nicola Scichilone, Giselda Colombo, Giorgio Walter Canonica, Giovanni Passalacqua, M D'Amato, Francesco Menzella, Vincenzo Patella, Francesca Puggioni, Roberta Parente, Paolo Solidoro, Giuseppe Spadaro, Sarah Barbuto, Erminia Ridolo, Elisabetta Favero, Matteo Bradicich, Marco Caminati, Gianna Camiciottoli, Gianenrico Senna, Heffler, Enrico, Blasi, Francesco, Latorre, Manuela, Menzella, Francesco, Paggiaro, Pierluigi, Pelaia, Girolamo, Senna, Gianenrico, Canonica, Giorgio Walter, Barbuto, Sarah, Bradicich, Matteo, Camiciottoli, Gianna, Caminati, Marco, Colombo, Giselda, Costantino, Maria Teresa, Crimi, Nunzio, Crivellaro, Mariangiola, D'Adda, Alice, D'Amato, Mariella, Favero, Elisabetta, Foschino, Maria Pia, Galeone, Carla, Guarnieri, Gabriella, Lombardi, Carlo, Parente, Roberta, Passalacqua, Giovanni, Patella, Vincenzo, Puggioni, Francesca, Ridolo, Erminia, Rolla, Giovanni, Savi, Eleonora, Scichilone, Nicola, Solidoro, Paolo, Spadaro, Giuseppe, Heffler E., Blasi F., Latorre M., Menzella F., Paggiaro P., Pelaia G., Senna G., Canonica G.W., Barbuto S., Bradicich M., Camiciottoli G., Caminati M., Colombo G., Costantino M.T., Crimi N., Crivellaro M., D'Adda A., D'Amato M., Favero E., Foschino M.P., Galeone C., Guarnieri G., Lombardi C., Parente R., Passalacqua G., Patella V., Puggioni F., Ridolo E., Rolla G., Savi E., Scichilone N., Solidoro P., and Spadaro G.

Subjects
Adult, Male, medicine.medical_specialty, Registry, Severe asthma, Exacerbation, Bronchiectasis, Comorbidities, Late-onset asthma, Nasal polyps, SANI, Immunology and Allergy, Omalizumab, Comorbidity, Settore MED/10 - Malattie Dell'Apparato Respiratorio, Atopy, 03 medical and health sciences, Bronchiectasi, 0302 clinical medicine, Food allergy, Internal medicine, Aged, Asthma, Eosinophils, Female, Humans, Immunoglobulin E, Italy, Middle Aged, Nasal Polyps, Registries, Rhinitis, Medicine, Bronchiectasis, Comorbidities, Late-onset asthma, Nasal polyps, Registry, SANI, Severe asthma, Immunology and Allergy, 030212 general & internal medicine, business.industry, Nasal polyp, medicine.disease, 030228 respiratory system, Asthma Control Questionnaire, Comorbiditie, business, Mepolizumab, medicine.drug
Abstract

Background Severe Asthma Network in Italy (SANI) is a registry of patients recruited by accredited centers on severe asthma. Objective To analyze epidemiological, clinical, inflammatory, functional, and treatment characteristics of severe asthmatics from the SANI registry. Methods All consecutive patients with severe asthma were included into the registry, without exclusion criteria to have real-life data on demographics, asthma control, treatments (including biologics), inflammatory biomarkers, and comorbidities. Results A total of 437 patients (mean age: 54.1 years, 57.2% females, 70.7% atopics, 94.5% in Global Initiative for Asthma severity step V) were enrolled into the study. The mean annual exacerbation rate was 3.75. The mean blood eosinophil level was 536.7 cells/mcL, and the average serum total IgE was 470.3 kU/L. Approximately 64% of patients were on regular oral corticosteroid treatment, 57% with omalizumab and 11.2% with mepolizumab. Most common comorbidities were rhinitis, nasal polyposis, and bronchiectasis. Patients with nasal polyposis had higher age of disease onset, higher blood eosinophil count, and lower frequency of atopy and atopic eczema. Bronchiectasis was associated with more frequent severe exacerbations, higher blood eosinophils, and total IgE. Stratifying patients, those with late-onset asthma were less frequently atopic (with less frequent allergic rhinitis and food allergy), and more frequently with nasal polyposis and higher serum total IgE levels. Conclusions This study revealed a high frequency of relevant comorbidities and that a substantial proportion of patients have late-onset asthma; all these features define specific different disease phenotypes. Severe asthma complexity and comorbidities require multidisciplinary approaches, led by specifically trained pulmonologists and allergists.

Published
2018

84. Mild/Moderate Asthma Network in Italy (MANI): a long-term observational study.

Author

Braido F, Blasi F, Canonica GW, Paggiaro P, Beghè B, Bonini M, Carpagnano GE, Del Giacco S, Lavorini F, Milanese M, Patella V, Santus P, Contoli M, Allegrini C, Baiardini I, Bonzano L, Caiaffa MF, Castelnuovo P, Corsico AG, Cosmi L, Costantino MT, Cottini M, Crimi N, Crivellaro MA, D'Alò S, Folletti I, Fornari D, Foschino-Barbaro MP, Franceschini L, Gargano D, Oliani KL, Maniscalco M, Melissari L, Montagni M, Montuschi P, Murgia N, Pannofino A, Papi A, Parente R, Pelaia G, Pini L, Puggioni F, Pulerà N, Resta O, Ricciardi L, Ridolo E, Savi E, Savoia F, Scala G, Senna G, Tripodi S, Vatrella A, Ventura MT, Viviano VM, and Yacoub MR

Subjects
Administration, Inhalation, Cross-Sectional Studies, Disease Progression, Humans, Prospective Studies, Quality of Life, Anti-Asthmatic Agents therapeutic use, Asthma drug therapy, Asthma epidemiology
Abstract

Objective: The prevalence of asthma in Italy is estimated to be around 4%; it affects approximately 2,000,000 citizens, and up to 80-90% of patients have mild-to-moderate asthma. Despite the clinical relevance of mild-to-moderate asthma, longitudinal observational data are very limited, including data on disease progression (worsening vs. improvement), the response to treatment, and prognosis. Studies are needed to develop long-term, observational, real-life research in large cohorts. The primary outcomes of this study will be based on prospective observation and the epidemiological evolution of mild and moderate asthma. Secondary outcomes will include patient-reported outcomes, treatments over time, disease-related functional and inflammatory patterns, and environmental and life-style influences., Methods: This study, called the Mild/Moderate Asthma Network of Italy (MANI), is a research initiative launched by the Italian Respiratory Society and the Italian Society of Allergology, Asthma and Clinical Immunology. MANI is a cluster-based, real world, cross-sectional, prospective, observational cohort study that includes 20,000 patients with mild-to-moderate asthma. (ClinicalTrials.gov Identifier: NCT04796844)., Results and Conclusion: Despite advances in asthma care, several research gaps remain to be addressed through clinical research. This study will add important new knowledge about long-term disease history, the transferability of clinical research results to daily practice, the efficacy of currently recommended strategies, and their impact on the burden and evolution of the disease., Abbreviations: MANI:Mild/Moderate Asthma Network of ItalySANI:Severe Asthma Network ItalyGINA:Global Initiative for AsthmaSABA:short acting β2-agonistsICS:inhaled corticosteroidsCRF:Case Report Form.

Published
2022
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