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1. 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, and Oliviero Rossi

Subjects
Quality of life, Atopic dermatitis, Allergic contact dermatitis, Hereditary angioedema, Cutaneous mastocytosis, Urticaria, Immunologic diseases. Allergy, RC581-607
Abstract

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
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2. Polyethylene Glycol and Polysorbate 80 Skin Tests in the Context of an Allergic Risk Assessment for Hypersensitivity Reactions to Anti-SARS-CoV-2 mRNA Vaccines

Author

Emanuele Nappi, Francesca Racca, Alessandra Piona, Maria Rita Messina, Sebastian Ferri, Donatella Lamacchia, Giuseppe Cataldo, Giovanni Costanzo, Lorenzo Del Moro, Francesca Puggioni, Giorgio Walter Canonica, Enrico Heffler, and Giovanni Paoletti

Subjects
SARS-CoV-2, vaccine, allergy, polyethylene glycol, polysorbate, skin tests, Medicine
Abstract

Concern has arisen about hypersensitivity reactions in patients with allergic reactions to drugs containing polyethylene glycol (PEG) or polysorbate 80 (PS80), excipients of currently available anti-SARS-CoV-2 mRNA vaccines. However, the actual utility of PEG and PS80 skin allergy testing is currently still debated. We retrospectively analyzed all cases of patients on whom we performed allergometric skin tests for PEG and PS80 in the context of a pre-vaccination screening (for patients with multiple hypersensitivity reactions to drugs for which these excipients were among the suspected agents) or following suspected hypersensitivity reactions to anti-SARS-CoV-2 vaccines. A total of 134 tests were performed for PEG and PS80, eight of which produced uninterpretable results (due to dermographism or non-specific reactions). Of the remaining 126 cases (85 pre-vaccinal and 41 post-vaccine reactions), 16 (12.7%) were positive for PEG and/or PS80. Stratifying by clinical indication, there were no statistically significant differences in the proportion of positive tests between patients evaluated in the context of the pre-vaccination screening and those evaluated after a vaccine reaction (10.6% vs. 17.1%, respectively, p = 0.306). Allergometric skin tests for PEG and PS80 in our case series resulted positive in an unexpectedly high proportion of patients, suggesting that testing for allergy to these two excipients should not be ignored in case of reasonable clinical suspicion.

Published
2023
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3. Allergy clinics in times of the SARS-CoV-2 pandemic: an integrated model

Author

Giacomo Malipiero, Enrico Heffler, Corrado Pelaia, Francesca Puggioni, Francesca Racca, Sebastian Ferri, Lina Spinello, Morena Merigo, Donatella Lamacchia, Giuseppe Cataldo, Melissa Sansonna, Giorgio Walter Canonica, and Giovanni Paoletti

Subjects
Allergy, Asthma, COVID-19, SARS-CoV-2, Pandemic, Immunotherapy, Immunologic diseases. Allergy, RC581-607
Abstract

Abstract Background Almost the entire World is experiencing the Coronavirus-Disease-2019 (COVID-19) pandemic, responsible, at the end of May 2020, of more than five million people infected worldwide and about 350,000 deaths. In this context, a deep reorganization of allergy clinics, in order to ensure proper diagnosis and care despite of social distancing measures expose, is needed. Main text The reorganization of allergy clinics should include programmed checks for severe and poorly controlled patients, application of digital medicine service for mild-to-moderate disease in well-controlled ones, postponement of non urgent diagnostic work-ups and domiciliation of therapies, whenever possible. As far as therapies, allergen immunotherapy (AIT) should not be stopped and sublingual immunotherapy (SLIT) fits perfectly for this purpose, since a drug home-delivery service can be activated for the entire pandemic duration. Moreover, biologic agents for severe asthma, chronic spontaneous urticaria and atopic dermatitis should be particularly encouraged to achieve best control possible of severe disease in times of COVID-19 and, whenever possible, home-delivery and self-administration should be the preferred choice. Conclusion During COVID-19 pandemic, allergists have the responsibility of balancing individual patients’ needs with public health issues, and innovative tools, such as telemedicine and digital medicine services, can be helpful to reduce the risk of viral spreading while delivering up-to-date personalized care.

Published
2020
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4. Successful SARS-CoV-2 vaccine allergy risk-management: The experience of a large Italian University Hospital

Author

Giovanni Paoletti, Francesca Racca, Alessandra Piona, Giulio Melone, Morena Merigo, Francesca Puggioni, Sebastian Ferri, Elena Azzolini, Michele Lagioia, Donatella Lamacchia, Giuseppe Cataldo, Maurizio Cecconi, Giorgio Walter Canonica, and Enrico Heffler

Subjects
Allergy, Anaphylaxis, COVID-19, Polyethylen glycole, Vaccine, Immunologic diseases. Allergy, RC581-607
Abstract

Background: Novel Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2) vaccines have been approved recently, and public concern regarding the risk of anaphylactic reactions arose after a few cases during the first days of mass vaccination. Polyethylene glycol (PEG) has been suggested as the most probable culprit agent for allergic reactions. Objective: We describe the allergy work-up protocol implemented for the vaccination campaign in our Center, aiming to allow the greatest number of people to be vaccinated safely. Methods: The protocol included the self-report of a history of suspected drug or vaccine allergies, and subsequent teleconsultation and allergometric tests for PEG and Polysorbate 80 (PS80). A desensitizing protocol of vaccine administration was applied to patients sensitized only to PS80, and to those with a suspect allergic reaction after the first vaccine dose. Results: 10.2% (414 out of 4042) of the entire vaccine population have been screened: only one patient resulted allergic to PEG and therefore excluded from the vaccination. Another patient was sensitized to PS80 only and safely vaccinated applying the desensitizing protocol. Seven subjects without a previous history of allergic disease experienced suspect hypersensitivity reactions to the first administered dose: one of them resulted allergic to PEG and was excluded from the second dose, while the others safely completed the vaccination with the desensitizing protocol. Conclusion: A careful allergological risk-assessment protocol significantly reduces the number of patients who would have avoided SARS-CoV-2 vaccination for their allergies and to effectively identify and manage those rare patients with sensitization to PEGs and/or PS80.

Published
2021
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5. Abstracts from the 10th C1-inhibitor deficiency workshop

Author

Alvin H. Schmaier, Marco Cicardi, Avner Reshef, Dumitru Moldovan, Attila Mócsai, Margarita López-Trascasa, Alberto López Lera, Nancy J. Brown, Anastasios E. Germenis, Rafael Filippelli-Silva, Diego A. Duarte, Renan P. Martin, Camila L. Veronez, Michel Bouvier, Michael Bader, Claudio M. Costa-Neto, João Bosco Pesquero, Xavier Charest-Morin, François Marceau, Georges-É. Rivard, Arnaud Bonnefoy, Éric Wagner, Márta L. Debreczeni, Zsuzsanna Németh, Erika Kajdácsi, Endre Schwaner, László Cervenak, Gábor Oroszlán, András Szilágyi, Ráhel Dani, Péter Závodszky, Péter Gál, József Dobó, Jacques Hébert, Matthieu Vincent, Jean-Nicolas Boursiquot, Hugo Chapdeleine, Marylin Desjardins, Benoit Laramée, Rémi Gagnon, Nancy Payette, Oleksandra Lepeshkina, Delphine Charignon, Arije Ghannam, Denise Ponard, Christian Drouet, Kusumam Joseph, Baby G. Tholanikunnel, Daniel J. Sexton, Allen P. Kaplan, Stefania Loffredo, Maria Bova, Anne Lise Ferrara, Angelica Petraroli, Chiara Suffritti, Nóra Veszeli, Andrea Zanichelli, Henriette Farkas, Gianni Marone, Samuel Luyasu, Bertrand Favier, Ludovic Martin, Kinga Viktória Kőhalmi, György Temesszentandrási, Katalin Várnai, Lilian Varga, Bruce L. Zuraw, Annette Feussner, Michael A. Tortorici, Dipti Pawaskar, Huamin Henry Li, John Anderson, Jonathan A. Bernstein, Ying Zhang, Ingo Pragst, on behalf of COMPACT investigators, Emel Aygören-Pürsün, Kraig Jacobson, Jim Christensen, Arthur Van Leerberghe, Yi Wang, Jennifer Schranz, Inmaculada Martinez-Saguer, Daniel Soteres, Urs Steiner, Vesna Grivcheva Panovska, William Rae, Werner Aberer, Aarnoud Huissoon, Anette Bygum, Markus Magerl, Jochen Graff, Hilary Longhurst, Ramón Lleonart, Lei Fang, Melanie Cornpropst, Desiree Clemons, Amanda Mathis, Phil Collis, Sylvia Dobo, William P. Sheridan, Marcus Maurer, Marc A. Riedl, Timothy Craig, Aleena Banerji, Mustafa Shennak, William Yang, Jovanna Baptista, Paula Busse, Ira Kalfus, Andrew McDonald, Shawn Qian, Anthony Roberts, Con Panousis, Tim Green, Andreas Gille, Maria Zamanakou, Gedeon Loules, Dorottya Csuka, Fotis Psarros, Faidra Parsopoulou, Matthaios Speletas, Davide Firinu, Tiziana Maria Angela De Pasquale, Alessandra Zoli, Anna Radice, Stefano Pizzimenti, Emmanouil Manoussakis, George N. Konstantinou, Valeria Bafunno, Vincenzo Montinaro, Mauro Cancian, Maurizio Margaglione, Konrad Bork, Karin Wulff, Guenther Witzke, Jochen Hardt, Laurence Bouillet, Teresa Caballero, Anete S. Grumach, Christelle Pommie, Irmgard Andresen, Carmen Escuriola Ettingshausen, Zeynep Gutowski, Karin Andritschke, Richard Linde, Noémi Andrási, Tamás Szilágyi, Iris Leibovich-Nassi, Christine Symons, John Dempster, Isabelle Boccon-Gibod, Anne Pagnier, Audrey Lehmann, Kristian B. Kreiberg, Sandra A. Nieto, Raquel Martins, Renata Martins, Alejandra Menendez, Solange O. R. Valle, Margarita Olivares, Maria E. Hernandez-Landeros, Elma Nievas, Natalia Fili, Olga M. Barrera, René Bailleau, Ana Maria Gallardo-Olivos, Masumi Grau, Julian Rodriguez-Galindo, Marlon J. O. Carabantes, Edison Zapata-Venegas, Mario Martinez Alfonso, Maria Rosario-Grauert, Manuel Ratti, Daniel Vaszquez, Dario Josviack, Luis Fernando Landivar-Salinas, Oscar M. E. Calderón-Llosa, Rolando Campilay-Sarmiento, Pablo Raby, Jose Fabiani, William R. Lumry, Henrike Feuersenger, Douglas J. Watson, Thomas Machnig, on behalf of the Investigators of the COMPACT study, Donatella Lamacchia, Adriana Hernanz, Ana Alvez, Mariana Lluncor, Maria Pedrosa, Rosario Cabañas, Nieves Prior, Patrik Nordenfelt, Mats Nilsson, Anders Lindfors, Carl-Fredrik Wahlgren, Janne Björkander, Roman Hakl, Pavel Kuklínek, Irena Krčmová, Jana Hanzlíková, Martina Vachová, Radana Zachová, Marta Sobotková, Jana Strenková, Jiří Litzman, Maria Palasopoulou, Gerasimina Tsinti, Panagiota Gianni, Maria Kompoti, Sofia Garrido, Wojciech Dyga, Anna Bogdali, Aleksander Obtułowicz, Mikolajczyk Tomasz, Ewa Czarnobilska, Krystyna Obtulowicz, Teofila Książek, Anna Koncz, Dominik Gulyás, Maria Staevska, Milos Jesenak, Katarina Hrubiskova, L. Bellizzi, A. Relan, Maddalena A. Wu, Antonio Castelli, Riccardo Colombo, Gianmarco Podda, Marta Del Medico, Emanuele Catena, Francesco Casella, Francesca Perego, Nada Afifi Afifi, Eleonora Tobaldini, Nicola Montano, for the IOS Study Group, Marta Sánchez-Jareño, Marcin Stobiecki, Krystyna Obtułowicz, Irina Guryanova, Ekaterina Polyakova, Viktar Lebedz, Andrej Salivonchik, Svetlana Aleshkevich, Mikhail Belevtsev, Melanie Nordmann-Kleiner, Susanne Trainotti, Janina Hahn, Jens Greve, Liudmyla Zabrodska, Maria L. Oliva Alonso, Rosangela P. Tórtora, Alfeu T. França, Marcia G. Ribeiro, Lisa Fu, Amin Kanani, Gina Lacuesta, Susan Waserman, Stephen Betschel, Melissa I. Espinosa, Francisco A. Contreras, Martin Hrubisko, Ludmila Vavrova, Peter Banovcin, Maryam Ayazi, Mohammad Reza Fazlollahi, Shiva Saghafi, Sajedeh Mohammadian, Susan Nabilou Deshiry, Kiana Bidad, Raheleh Shokouhi Shoormasti, Iraj Mohammadzadeh, Mohammad Hassan Bemanian, Seyed Alireza Mahdaviani, Zahra Pourpak, Anna Valerieva, Mariela Vasileva, Tsvetelina Velikova, Elena Petkova, Vasil Dimitrov, Ruggero Di Maulo, on behalf of participating centers, Raz Somech, Hava Golander, Erika J. Sifuentes, Catherine Mansard, Anne Gompel, Bernard Floccard, Claire Blanchard-Delaunay, David Launay, Olivier Fain, Alain Sobel, Stéphane Gayet, Stéphanie Amarger, Guillaume Armengol, Yann Ollivier, Ariane Zélinsky-Gurung, Pierre-Yves Jeandel, Gisèle Kanny, Brigitte Coppéré, Marie Dubrel, Fabien Pelletier, Aurélie Du Thanh, Sébastien Trouiller, Jérôme Laurent, Claire De Moreuil, Christine Audouin Pajot, Alexandre Belot, Ana Rodríguez, Dasha Roa, Alicia Prieto, Maria Luisa Baeza, Borislava Krusheva, Stephanie K. A. Almeida, Rosemeire N. Constantino-Silva, Nyla Melo, Joanna Araujo Simoes, Sandra Mitie U. Palma, Jane da Silva, Bruna F. de Azevedo, Eli Mansour, Teresa González-Quevedo, Carmen Marcos, Teófilo Lobera, Blanca Sáenz de San Pedro, Ernie Avilla, Jacquie Badiou, Karen Binkley, Rozita Borici-Mazi, Linda Howlett, Paul K. Keith, Anne Rowe, Peter Waite, Aurore Billebeau, Isabelle Boccon-Gibbod, Kristina Lis, Yael Laitman, Eitan Friedman, N. M. Gokmen, O. Gulbahar, H. Onay, Z. P. Koc, and A. Z. Sin

Subjects
Immunologic diseases. Allergy, RC581-607
Published
2017
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6. Successful SARS-CoV-2 vaccine allergy risk-management: the experience of a large Italian University Hospital

Author

Morena Merigo, Michele Lagioia, Giorgio Walter Canonica, Giuseppe Cataldo, Francesca Puggioni, Donatella Lamacchia, Giulio Melone, Francesca Racca, Maurizio Cecconi, Enrico Heffler, Alessandra Piona, Elena Azzolini, Sebastian Ferri, and Giovanni Paoletti

Subjects
Pulmonary and Respiratory Medicine, Drug, Pediatrics, medicine.medical_specialty, Allergy, media_common.quotation_subject, Immunology, Population, SIAAIC, Italian Society of Allergology Asthma and Clinical Immunology, Disease, Culprit, DMG, Dimyristoyl glycerol, Article, medicine, CDC, Center for Diseases Control, Immunology and Allergy, education, Anaphylaxis, Sensitization, media_common, education.field_of_study, DPT, Diphteria Pertussis Tetanus, AAIITO, Italian Territorial and Hospital Allergologists and Immunologists Association, business.industry, COVID-19, RC581-607, medicine.disease, FDA, Food and Drug Administration, Vaccination, COVID-19, Coronavirus-Disease-2019, medicine.anatomical_structure, SARS-CoV-2, Severe Acute Respiratory Syndrome Coronavirus 2, PEG, Polyethylene glycol, PS80, Polysorbate 80, Immunologic diseases. Allergy, business, Polyethylen glycole, Vaccine
Abstract

Background Novel Severe Acute Respiratory Syndrome Coronavirus 2 (SARS-CoV-2) vaccines have been approved recently, and public concern regarding the risk of anaphylactic reactions arose after a few cases during the first days of mass vaccination. Polyethylene glycol (PEG) has been suggested as the most probable culprit agent for allergic reactions. Objective We describe the allergy work-up protocol implemented for the vaccination campaign in our Center, aiming to allow the greatest number of people to be vaccinated safely. Methods The protocol included the self-report of a history of suspected drug or vaccine allergies, and subsequent teleconsultation and allergometric tests for PEG and Polysorbate 80 (PS80). A desensitizing protocol of vaccine administration was applied to patients sensitized only to PS80, and to those with a suspect allergic reaction after the first vaccine dose. Results 10.2% (414 out of 4042) of the entire vaccine population have been screened: only one patient resulted allergic to PEG and therefore excluded from the vaccination. Another patient was sensitized to PS80 only and safely vaccinated applying the desensitizing protocol. Seven subjects without a previous history of allergic disease experienced suspect hypersensitivity reactions to the first administered dose: one of them resulted allergic to PEG and was excluded from the second dose, while the others safely completed the vaccination with the desensitizing protocol. Conclusion A careful allergological risk-assessment protocol significantly reduces the number of patients who would have avoided SARS-CoV-2 vaccination for their allergies and to effectively identify and manage those rare patients with sensitization to PEGs and/or PS80.

Published
2021

7. Orofacial granulomatosis: Clinical and therapeutic features in an Italian cohort and review of the literature

Author

Mauro Cancian, Stefania Loffredo, Giovanni Pellacani, Giovanni Rolla, Amato de Paulis, Filomena Maio, Luigi Giovanni Cremonte, Massimo Triggiani, Gianfranco Vitiello, Roberta Parente, Paola Parronchi, Riccardo Senter, Giuseppe Spadaro, Davide Firinu, Eustachio Nettis, Laura Bonzano, Maria Bova, Donatella Lamacchia, Elisa Boni, Francesco Arcoleo, Angelica Petraroli, Stefano Del Giacco, Luisa Brussino, Aikaterini Detoraki, Maria Rosaria Galdiero, Galdiero, M. R., Maio, F., Arcoleo, F., Boni, E., Bonzano, L., Brussino, L., Cancian, M., Cremonte, L., Del Giacco, S. R., De Paulis, A., Detoraki, A., Firinu, D., Lamacchia, D., Loffredo, S., Nettis, E., Parente, R., Parronchi, P., Pellacani, G., Petraroli, A., Rolla, G., Senter, R., Triggiani, M., Vitiello, G., Spadaro, G., and Bova, M.

Subjects
0301 basic medicine, medicine.medical_specialty, Delayed Diagnosis, Immunology, orofacial granulomatosis, granulomatous cheiliti, Inflammatory bowel disease, Granulomatosis, Orofacial, Atopy, 03 medical and health sciences, 0302 clinical medicine, orofacial granulomatosi, granulomatous cheilitis, Italian registries, Melkersson-Rosenthal syndrome, Cervical lymphadenopathy, Melkersson–Rosenthal syndrome, medicine, Immunology and Allergy, Humans, Granulomatosis, Orofacial, Tumor Necrosis Factor Inhibitor, Italy, Tumor Necrosis Factor Inhibitors, Melkersson-Rosenthal Syndrome, business.industry, Delayed Diagnosi, Soft tissue, medicine.disease, Dermatology, 030104 developmental biology, 030228 respiratory system, Cohort, Italian registrie, Orofacial granulomatosis, Sarcoidosis, medicine.symptom, business, Human
Abstract

Background Orofacial granulomatosis (OFG) is characterized by granulomatous inflammation of the soft tissues of maxillofacial region. We explored OFG patients from 10 different Italian centers and summarized the most recent literature data. Methods A review of patients with OFG was carried out. An extensive online literature search was performed to identify studies reporting diagnosis and management of OFG. Results Thirty-nine patients were recruited between January 2018 and February 2020. Most of them (97.4%) displayed involvement of the lips, and 28.2% suffered from Melkersson-Rosenthal syndrome. Two patients received diagnosis of CD and one patient of sarcoidosis, suggesting secondary OFG. Oral aphthosis and cervical lymphadenopathy were also described. The mean diagnostic delay was 3.4 years. Histological evaluation was performed in 34/39 patients (87.2%); non-caseating granulomas were found in 73.5% of them. Neurological symptoms (28.2%), gastrointestinal symptoms in absence of overt inflammatory bowel disease (IBD) (20.5%), and atopy (35.9%) were also identified. Therapeutic approaches varied among the centers. Steroids (51.3%) were used with good or partial results. Anti-TNF-α and anti-IgE monoclonal antibodies were used in 6 (15.4%) and 1 (2.6%) patients, respectively, with variable results. Surgery was the choice for 2 patients with good response. Conclusions OFG is a rare and neglected disease showing multiple clinical phenotypes. While early diagnosis is crucial, management is difficult and highly dependent on the expertise of clinicians due to the lack of international guidelines. There is a need to establish registry databases and address challenges of long-term management.

Published
2021

8. Corrigendum to 'Rapid infusions of human normal immunoglobulin 50g/l are safe and well tolerated in immunodeficiencies and immune thrombocytopenia' [Int. Immunopharmacol. 44 (2017) 38-42]

Author

Antonio Pecoraro, Liliana Nappi, Serena Bongiovanni, Giuseppe Spadaro, Elisabet Speckman, Jan Janssen, Donatella Lamacchia, Alberto Bosi, Valentina Carrai, Cinzia Milito, Francesca Bacchiarri, Andrea Ferraro, Dietmar Reichert, Isabella Quinti, Andrea Matucci, Chiara Guarnieri, Azra Hibbeler, Alessandra Vultaggio, Spadaro, Giuseppe, Vultaggio, Alessandra, Bosi, Alberto, Reichert, Dietmar, Janssen, Jan, Lamacchia, Donatella, Nappi, Liliana, Pecoraro, Antonio, Milito, Cinzia, Ferraro, Andrea, Matucci, Andrea, Bacchiarri, Francesca, Carrai, Valentina, Hibbeler, Azra, Speckman, Elisabet, Guarnieri, Chiara, Bongiovanni, Serena, and Quinti, Isabella

Subjects
Pharmacology, business.industry, INT, Immunology, Human Normal Immunoglobulin, Medicine, Immunology and Allergy, business, Immune thrombocytopenia
Published
2017

9. Abstracts from the 10th C1-inhibitor deficiency workshop

Author

Mikhail Belevtsev, Jens Greve, Pierre-Yves Jeandel, Ana Maria Gallardo-Olivos, William R. Lumry, Yael Laitman, Marc A. Riedl, Jean-Nicolas Boursiquot, Dasha Roa, Péter Závodszky, Kraig Jacobson, Susanne Trainotti, Maria Staevska, Márcia Gonçalves Ribeiro, Zeynep Gutowski, François Marceau, Alejandra Menendez, Eric Wagner, Y. Ollivier, Alvin H. Schmaier, Rolando Campilay-Sarmiento, Amin S. Kanani, Valeria Bafunno, Christine Symons, Svetlana Aleshkevich, Stephanie K. A. Almeida, Kusumam Joseph, Teresa Caballero, Hava Golander, Nancy J. Brown, Xavier Charest-Morin, Peter Banovcin, Arnaud Bonnefoy, Sajedeh Mohammadian, Mustafa Shennak, William P. Sheridan, Ira Kalfus, Claudio M. Costa-Neto, Rafael Filippelli-Silva, Anete Sevciovic Grumach, Solange Oliveira Rodrigues Valle, Oscar M. E. Calderón-Llosa, Panagiota Gianni, Emel Aygören-Pürsün, Rosemeire Navickas Constantino-Silva, Zsuzsanna Németh, Irmgard Andresen, Konrad Bork, Mauro Cancian, Claire de Moreuil, Janne Björkander, Melissa I. Espinosa, Tamás Szilágyi, Maria Rosario-Grauert, Jane da Silva, Michael Bader, Ying Zhang, Jana Hanzlíková, Sylvia Dobo, Carl-Fredrik Wahlgren, Jacquie Badiou, Diego A. Duarte, Marta Del Medico, Stefania Loffredo, Avner Reshef, Maria Palasopoulou, Ludmila Vavrova, Marie Dubrel, Raheleh Shokouhi Shoormasti, Bruce L. Zuraw, Susan Nabilou Deshiry, Shiva Saghafi, Ekaterina Polyakova, Matthieu Vincent, Douglas J. Watson, Mohammad Reza Fazlollahi, Anne Rowe, Shawn Qian, Blanca Sáenz de San Pedro, Christelle Pommie, Sofía Garrido, Sandra Mitie Ueda Palma, András Szilágyi, Luis Fernando Landivar-Salinas, Audrey Lehmann, Maria L. Baeza, Kiana Bidad, Hugo Chapdeleine, Stéphane Gayet, Davide Firinu, Nancy Payette, Christine Pajot, Nyla Melo, Pavel Kuklínek, Pablo Raby, Nóra Veszeli, Ingo Pragst, Desiree Clemons, Anne Pagnier, Attila Mócsai, Carmen Escuriola Ettingshausen, Henrike Feuersenger, Noémi Andrási, Paul K. Keith, Phil Collis, Ernie Avilla, Olivier Fain, Iris Leibovich-Nassi, Dario O. Josviack, Jiří Litzman, Con Panousis, Denise Ponard, Sébastien Trouiller, Ráhel Dani, Francesco Casella, Ana Rodríguez, Isabelle Boccon-Gibod, Nada Afifi Afifi, Olga M. Barrera, Gina Lacuesta, Gian Marco Podda, Anne Lise Ferrara, Samuel Luyasu, Ludovic Martin, Patrik Nordenfelt, Linda Howlett, William H. Yang, Sandra A. Nieto, Anders Lindfors, Zahra Pourpak, Stéphanie Amarger, Adriana Hernanz, Jochen Hardt, Julian Rodriguez-Galindo, Claire Blanchard-Delaunay, Antonio Castelli, Marta Sánchez-Jareño, Renata Martins, Bertrand Favier, Yi Wang, Rosario Cabañas, Anthony Roberts, Renan Paulo Martin, Alexandre Belot, Vincenzo Montinaro, Eitan Friedman, João Bosco Pesquero, Maria Kompoti, Bruna Franca Azevedo, Mikolajczyk Tomasz, Georges-É. Rivard, Katalin Várnai, Gábor Oroszlán, Arije Ghannam, David Launay, Fotis Psarros, Manuel Ratti, Maria Luiza Oliva Alonso, Bernard Floccard, Mariana Lluncor, Karen Binkley, Gianni Marone, Anne Gompel, Dominik Gulyás, John Dempster, Liudmyla Zabrodska, Emanuele Catena, Anna Bogdali, Irina Guryanova, Andrej Salivonchik, Camila Lopes Veronez, L. Fang, Eleonora Tobaldini, Lilian Varga, Chiara Suffritti, Daniel Vaszquez, Peter Waite, Maria Zamanakou, Ana Alvez, Laurence Bouillet, Péter Gál, Susan Waserman, Krystyna Obtułowicz, Marcin Stobiecki, Alain Sobel, Karin Wulff, A. Z. Sin, Margarita Olivares, Dipti Pawaskar, Mats Nilsson, Endre Schwaner, Brigitte Coppere, Anna Valerieva, Remi Gagnon, Marlon J. O. Carabantes, Stefano Pizzimenti, H. Onay, Paula J. Busse, Delphine Charignon, William Rae, Marco Cicardi, Kinga Viktória Kőhalmi, Inmaculada Martinez-Saguer, Daniel J. Sexton, Matthaios Speletas, Aarnoud Huissoon, René Bailleau, Fabien Pelletier, Elena Petkova, Joanna Araujo Simoes, Guillaume Armengol, Amanda Mathis, Rosangela P. Tortora, Raz Somech, Andreas Gille, Anastasios E. Germenis, Martin Hrubisko, Marylin Desjardins, Dorottya Csuka, María Pedrosa, Jose Fabiani, Stephen Betschel, Timothy J. Craig, Vasil Dimitrov, Andrew McDonald, Alberto López Lera, Márta L. Debreczeni, Eli Mansour, Teofila Książek, Teófilo Lobera, Rozita Borici-Mazi, O. Gulbahar, Aleena Banerji, Jochen Graff, Marta Sobotkova, Annette Feussner, Andrea Zanichelli, Ewa Czarnobilska, Anna Koncz, Milos Jesenak, Edison Zapata-Venegas, Aurore Billebeau, Gedeon Loules, Melanie Nordmann-Kleiner, Werner Aberer, Richard Linde, Guenther Witzke, Kristina Lis, Markus Magerl, Jana Strenková, Emmanouil Manoussakis, József Dobó, Viktar Lebedz, Masumi Grau, Raquel Martins, Melanie Cornpropst, Jovanna Baptista, Marcus Maurer, Dumitru Moldovan, György Temesszentandrási, Isabelle Boccon-Gibbod, Thomas Machnig, C Marcos, Erika Kajdácsi, Tim Green, John Anderson, Karin Andritschke, D. Soteres, Riccardo Colombo, Mario Martinez Alfonso, Anna Radice, Huamin Henry Li, Tsvetelina Velikova, Ruggero Di Maulo, Mariela Borisova Vasileva, Angelica Petraroli, Francesca Perego, Francisco A. Contreras, Roman Hakl, Urs C. Steiner, Anurag Relan, N. Prior, Irena Krčmová, Jennifer Schranz, Martina Vachová, Baby G. Tholanikunnel, R. Lleonart, Maria Bova, Allen P. Kaplan, N. M. Gokmen, Nicola Montano, Margarita López-Trascasa, Tiziana Maria Angela De Pasquale, Alicia Prieto, Kristian Buur Kreiberg, Mohammad Hassan Bemanian, Alessandra Zoli, Michael A. Tortorici, Erika J. Sifuentes, László Cervenak, Borislava Krusheva, Maria E. Hernandez-Landeros, Wojciech Dyga, Oleksandra Lepeshkina, Elma Nievas, L. Bellizzi, Christian Drouet, C. Mansard, Michel Bouvier, Aurélie Du Thanh, Donatella Lamacchia, Janina Hahn, Radana Zachova, Iraj Mohammadzadeh, T González-Quevedo, Z. P. Koc, Jacques Hébert, Hilary Longhurst, Maddalena Alessandra Wu, Ariane Zélinsky-Gurung, Jim Christensen, Maurizio Margaglione, Maryam Ayazi, Vesna Grivcheva Panovska, Faidra Parsopoulou, Jonathan A. Bernstein, Anette Bygum, Seyed Alireza Mahdaviani, Henriette Farkas, Aleksander Obtułowicz, Natalia Fili, Gisèle Kanny, Gerasimina Tsinti, Alfeu Tavares França, George N. Konstantinou, Benoit Laramée, Katarina Hrubiskova, Lisa Fu, J. Laurent, and Arthur Van Leerberghe

Subjects
lcsh:Immunologic diseases. Allergy, 0301 basic medicine, 03 medical and health sciences, 030102 biochemistry & molecular biology, C1 inhibitor deficiency, business.industry, Immunology, Medicine, General Medicine, lcsh:RC581-607, business, Meeting Abstracts
Published
2017

10. Rapid infusions of human normal immunoglobulin 50g/l are safe and well tolerated in immunodeficiencies and immune thrombocytopenia

Author

Chiara Guarnieri, Jan Janssen, Isabella Quinti, Cinzia Milito, Alberto Bosi, Andrea Matucci, Donatella Lamacchia, Andrea Ferraro, Liliana Nappi, Serena Bongiovanni, Francesca Bacchiarri, Antonio Pecoraro, Giuseppe Spadaro, Valentina Carrai, Elisabet Speckman, Dietmar Reichert, Alessandra Vultaggio, Azra Hibbeler, Spadaro, Giuseppe, Vultaggio, Alessandra, Alberto Bosi, A., Reichert, Dietmar, Janssen, Jan, Lamacchia, Donatella, Nappi, Liliana, Pecoraro, Antonio, Milito, Cinzia, Ferraro, Andrea, Matucci, Andrea, Bacchiarri, Francesca, Carrai, Valentina, Hibbeler, Azra, Speckman, Elisabet, Guarnieri, Chiara, Bongiovanni, Serena, and Quinti, Isabella

Subjects
0301 basic medicine, Tachycardia, Male, Pediatrics, Cost effectiveness, Intravenous immunoglobulin, Quality of life, Rapid infusion, Adolescent, Adult, Female, Humans, Immunoglobulins, Intravenous, Immunologic Deficiency Syndromes, Immunotherapy, Middle Aged, Patient Compliance, Prospective Studies, Purpura, Thrombocytopenic, Idiopathic, Quality of Life, Treatment Outcome, Young Adult, Immunology and Allergy, Immunology, Pharmacology, 0302 clinical medicine, hemic and lymphatic diseases, Cost effectivene, Young adult, Prospective cohort study, Immunologic Deficiency Syndrome, Idiopathic, Tolerability, medicine.symptom, Intravenous, Human, medicine.medical_specialty, Vital signs, Immunoglobulins, 03 medical and health sciences, medicine, Adverse effect, Purpura, business.industry, Thrombocytopenic, Prospective Studie, 030104 developmental biology, Blood pressure, Immunoglobulins, Intravenou, business, 030215 immunology
Abstract

Intravenous immunoglobulin (IVIg) is accepted as an effective and well-tolerated treatment for primary and secondary immunodeficiencies (ID) and immune thrombocytopenia (ITP). Adverse reactions of IVIg are usually mild, comprising transient flu-like symptoms, change in blood pressure and tachycardia. However IVIg therapy can be burdensome for both patients and healthcare facilities, since the infusion may take up to 4 h to administer. The objective of our multicentre, prospective, open-label phase III trial was to evaluate the tolerability and safety of human normal immunoglobulin 50 g/l (Ig VENA) at high intravenous infusion rates in adult patients with ID and ITP who had previously tolerated IVIg treatment, by progressively increasing infusion rate up to 8 ml/kg/hr. 39 ID patients received three infusions, 5 ITP patients received up to a maximum of 5 infusions for a maximum of 5 days. Overall 55 adverse events were reported in 18 patients, and all were mild and self-limiting. Two serious adverse events occurred in ID patients and 1 in an ITP patient; none was fatal or treatment-related. No clinically significant changes or abnormalities were observed in vital signs, laboratory results and HRQoL. In summary, in this study, more rapid IVIg infusions were well tolerated by ID and ITP patients, while maintaining their quality of life, helping to minimise the time spent in outpatient hospital visiting to potentially optimise adherence to treatment.

Published
2016

11. Determinants of health related quality of life (HRQoL) in adult patients with hereditary angioedema due to C1 inhibitor deficiency (C1-INH-HAE)

Author

Adriana Hernanz, Teresa Caballero, Rosario Cabañas, Marina Lluncor, N. Prior, María Pedrosa, Ana Alvez, and Donatella Lamacchia

Subjects
Pediatrics, medicine.medical_specialty, Adult patients, Quality of life, C1 inhibitor deficiency, business.industry, Immunology, Hereditary angioedema, medicine, Immunology and Allergy, Social determinants of health, business, medicine.disease
Published
2018

12. FRI0035 Expression and functions of vascular endothelial growth factor and angiopoietin receptors in human synovial mast cells

Author

Donatella Lamacchia, Nella Prevete, Felice Rivellese, C. Pelosi, Gianni Marone, F.W. Rossi, and A de Paulis

Subjects
Mast cell chemotaxis, medicine.medical_treatment, Immunology, Stem cell factor, Biology, medicine.disease, Mast cell, General Biochemistry, Genetics and Molecular Biology, Interleukin 33, chemistry.chemical_compound, medicine.anatomical_structure, Cytokine, Rheumatology, chemistry, Synovitis, medicine, Cancer research, Immunology and Allergy, Tumor necrosis factor alpha, Histamine
Abstract

Background Mast cells accumulate in the synovial tissue of patients with rheumatoid arthritis (RA) and several data in murine models demonstrate that they can contribute importantly to the initiation of inflammatory arthritis. In fact, mast cell activation in the rheumatoid lesion provides numerous mediators, including histamine, heparin, proteinases, leukotrienes and multifunctional cytokines, that contribute to inflammatory and degradative processes, especially at sites of cartilage erosion. Little is known, however, about the factors responsible for mast cells accumulation in inflamed synovia. It is probable that Stem Cell Factor (SCF), the signature mast cell growth and differentiation factor and potent mast cell chemotaxin, plays a central role in regulating synovial mast cell trafficking. Indeed, the typical arthritic cytokine TNF induces expression and release of SCF by synovial fibroblasts, promoting mast cell chemotaxis. Among the other possible mediators, it is known that angiogenic factors are highly expressed in synovial fluids and sera of RA patients. Previous experimental data from our group support the hypothesis that vascular endothelial growth factors (VEGFs), although classically considered relatively endothelial-cell-specific factors, can activate immune cells through their receptors. Therefore we hypothesized that VEGF and angiopoietins could be responsible for mast cells accumulation in synovia. Objectives We aimed to characterize the expression and functions of angiogenic receptors on human synovial mast cells (HSyMCs) to identify a non-immunological activation pattern implicated in mast cells recruitment in synovial tissue. Methods We evaluated the expression of vascular endothelial growth factor receptors 1 and 2 (VEGFR-1 and VEGFR-2) and angiopoietin receptors (Tie1 and Tie2) mRNAs in human mast cells lines (LAD-2 and HMC-1) by RT-PCR. The surface expression of the receptors was then confirmed by Flow Cytometry on HSyMCs isolated by enzymatic digestion from synovial tissue obtained from patients undergoing synovectomy. Finally, we evaluated the effects of angiogenic factors on HSyMC chemotaxis using a Boyden chamber. Results Human mast cells lines (LAD-2 and HMC-1) constitutively express mRNAs for VEGFR-1, VEGFR-2, Tie1 and Tie2. HSyMCs constitutively express on their surface VEGFR-1, VEGFR-2, Tie1 and Tie2. VEGF-A 165 (50–500 ng/ml) induced HSyMCs chemotaxis with a potency similar to SCF. The chemotactic effects were mediated by the activation of both VEGFR-1 and VEGFR-2, as demonstrated by blocking experiments. We also observed that Ang1, but not Ang2, induced migration of HSyMCs through the engagement of Tie2. Conclusions Our data indicate that HSyMCs are a target of angiogenic factors, which, in addition to the specific activation of endothelial cells, might play a wider role in inflammatory response in RA synovitis by activating immune cells. Here we also identify a non-immunological mechanism that accounts for the chemotactic activation of human synovial mast cells. References de Paulis A, Prevete N, Fiorentino I, et al. Expression and functions of the vascular endothelial growth factors and their receptors in human basophils. J Immunol 2006;177:7322-31 Nigrovic PA, Lee DM. Synovial mast cells: role in acute and chronic arthritis. Immunol Rev 2007;217:19-37 Disclosure of Interest None Declared

Published
2013

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