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1. 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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2. Hereditary angioedema: Assessing the hypothesis for underlying autonomic dysfunction.

Author

Maddalena A Wu, Francesco Casella, Francesca Perego, Chiara Suffritti, Nada Afifi Afifi, Eleonora Tobaldini, Andrea Zanichelli, Chiara Cogliati, Nicola Montano, and Marco Cicardi

Subjects
Medicine, Science
Abstract

Attacks of Hereditary Angioedema due to C1-inhibitor deficiency (C1-INH-HAE)are often triggered by stressful events/hormonal changes.Our study evaluates the relationship between autonomic nervous system (ANS) and contact/complement system activation.Twenty-three HAE patients (6 males, mean age 47.5±11.4 years) during remission and 24 healthy controls (8 males, mean age 45.3±10.6 years) were studied. ECG, beat-by-beat blood pressure, respiratory activity were continuously recorded during rest (10') and 75-degrees-head-up tilt (10'). C1-INH, C4, cleaved high molecular weight kininogen (cHK) were assessed; in 16 patients and 11 controls plasma catecholamines were also evaluated. Spectral analysis of heart rate variability allowed extraction of low-(LF) and high-(HF) frequency components, markers of sympathetic and vagal modulation respectively.HAE patients showed higher mean systolic arterial pressure (SAP) than controls during both rest and tilt. Tilt induced a significant increase in SAP and its variability only in controls. Although sympathetic modulation (LFnu) increased significantly with tilt in both groups, LF/HF ratio, index of sympathovagal balance, increased significantly only in controls. At rest HAE patients showed higher noradrenaline values (301.4±132.9 pg/ml vs 210.5±89.6pg/ml, p = 0.05). Moreover, in patients tilt was associated with a significant increase in cHK, marker of contact system activation (49.5 ± 7.5% after T vs 47.1 ± 7.8% at R, p = 0.01).Our data are consistent with altered ANS modulation in HAE patients, i.e. increased sympathetic activation at rest and blunted response to orthostatic challenge. Tilt test-induced increased HK cleavage suggests a link between stress and bradykinin production.

Published
2017
Full Text
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3. 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

4. Hereditary angioedema: Assessing the hypothesis for underlying autonomic dysfunction

Author

Eleonora Tobaldini, Nada Afifi Afifi, Maddalena Alessandra Wu, Chiara Suffritti, Marco Cicardi, Francesco Casella, Chiara Cogliati, Andrea Zanichelli, Nicola Montano, and Francesca Perego

Subjects
Male, Physiology, Vascular Permeability, Social Sciences, lcsh:Medicine, Blood Pressure, 030204 cardiovascular system & hematology, Pathology and Laboratory Medicine, Vascular Medicine, Nervous System, Epithelium, Orthostatic vital signs, chemistry.chemical_compound, Electrocardiography, 0302 clinical medicine, Heart Rate, Animal Cells, Blood plasma, Medicine and Health Sciences, Heart rate variability, Psychology, lcsh:Science, Immune Response, Multidisciplinary, Middle Aged, Body Fluids, Blood, Hereditary angioedema, Female, Anatomy, Cellular Types, Research Article, Adult, medicine.medical_specialty, Immunology, Cardiology, Bradykinin, Psychological Stress, Autonomic Nervous System, Blood Plasma, 03 medical and health sciences, Signs and Symptoms, Diagnostic Medicine, Internal medicine, Heart rate, Mental Health and Psychiatry, medicine, Humans, Inflammation, business.industry, lcsh:R, Angioedemas, Hereditary, Biology and Life Sciences, Endothelial Cells, Epithelial Cells, Cell Biology, medicine.disease, Autonomic nervous system, Endocrinology, Blood pressure, Biological Tissue, chemistry, Case-Control Studies, lcsh:Q, business, 030217 neurology & neurosurgery
Abstract

Background Attacks of Hereditary Angioedema due to C1-inhibitor deficiency (C1-INH-HAE)are often triggered by stressful events/hormonal changes. Objective Our study evaluates the relationship between autonomic nervous system (ANS) and contact/complement system activation. Methods Twenty-three HAE patients (6 males, mean age 47.5±11.4 years) during remission and 24 healthy controls (8 males, mean age 45.3±10.6 years) were studied. ECG, beat-by-beat blood pressure, respiratory activity were continuously recorded during rest (10') and 75-degrees-head-up tilt (10'). C1-INH, C4, cleaved high molecular weight kininogen (cHK) were assessed; in 16 patients and 11 controls plasma catecholamines were also evaluated. Spectral analysis of heart rate variability allowed extraction of low-(LF) and high-(HF) frequency components, markers of sympathetic and vagal modulation respectively. Results HAE patients showed higher mean systolic arterial pressure (SAP) than controls during both rest and tilt. Tilt induced a significant increase in SAP and its variability only in controls. Although sympathetic modulation (LFnu) increased significantly with tilt in both groups, LF/HF ratio, index of sympathovagal balance, increased significantly only in controls. At rest HAE patients showed higher noradrenaline values (301.4±132.9 pg/ml vs 210.5±89.6pg/ml, p = 0.05). Moreover, in patients tilt was associated with a significant increase in cHK, marker of contact system activation (49.5 ± 7.5% after T vs 47.1 ± 7.8% at R, p = 0.01). Conclusions Our data are consistent with altered ANS modulation in HAE patients, i.e. increased sympathetic activation at rest and blunted response to orthostatic challenge. Tilt test-induced increased HK cleavage suggests a link between stress and bradykinin production.

Published
2017

5. Autonomic Modulation and Contact System in Patients with Hereditary Angioedema Due to C1 Inhibitor Deficiency

Author

Eleonora Tobaldini, Nicola Montano, Nada Afifi Afifi, Chiara Suffritti, Marco Cicardi, Antonio Gidaro, Maddalena Alessandra Wu, Francesca Perego, Andrea Zanichelli, and Liliane Appratto de Souza

Subjects
C1 inhibitor deficiency, business.industry, Immunology, Contact system, Hereditary angioedema, Immunology and Allergy, Medicine, In patient, Autonomic modulation, business, medicine.disease
Published
2017

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