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1. Benefits of a Juvenile Arthritis Support Program (JASP-1) for children recently diagnosed with Juvenile Idiopathic Arthritis and their parents

Author

Karina Mördrup, Johanna Granhagen Jungner, Eva Broström, Karin Palmblad, and Cecilia Bartholdson

Subjects
JIA, Support program, Patient satisfaction, Patient outcome assessment, Diseases of the musculoskeletal system, RC925-935
Abstract

Abstract Background Medical treatment for children with Juvenile Idiopathic Arthritis (JIA) has improved radically since the development of biological disease-modifying antirheumatic drugs. However, children suffer from pain and anxiety, and parents often experience loneliness and lack of support. Some parents reported that information provided at the time their child was diagnosed could be difficult to assimilate. Therefore, the aim of this study was to develop a Juvenile Arthritis Support Program (JASP-1) for children recently diagnosed with JIA and their parents. Moreover, the aim was to explore patients´ and parents´ experiences with JASP-1 and its potential impact on patients´ physical health. Methods JASP-1 included seven patient- and family-centered clinical visit from time of diagnose and one year ahead. Data were collected from a study-specific questionnaire answered by children and their parents after participation in JASP-1 and from the pediatric rheumatology register. The study-specific questionnaire explored participants´ experience with the care they received during their first year with JIA. Registry and questionnaire data from the intervention (JASP-1) group was compared to a control group. Results The analysis revealed that children and parents who completed JASP-1 were more satisfied with the care they had received during their first year with JIA than the control group. The results also showed that children who completed JASP-1 were assessed as having better overall health after 12 months, than children in the control group (JASP-1 = mean 4.33, 95% Confidence Interval (CI) 4.17 − 4.46), (Control = mean 3.68, 95% CI 3.29 − 4.06), (p = 0.002). Moreover, children in the JASP-1 group had less disease impact on daily life (JASP-1 = mean 0.15, 95% CI 0.07 − 0.24) (Control = mean 0.40, 95% CI 0.13 − 0.67), (p = 0.017) and less active joints than the control group (JASP-1 = mean 0.62, 95% CI 0.35 − 1.58), (Control = mean 0.87, 95% CI 0.18 − 1.56), (p = 0.054). Conclusion A support program like JASP-1 could be an effective way of not only supporting children newly diagnosed with JIA and their parents psychologically but may also increase children’s overall physical health and improve quality of care within pediatric rheumatology. Trial registration Retrospectively registered in ClinicalTrials.gov, the 13th of February with ID NCT06284616.

Published
2024
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3. Immunoprofiling of active and inactive systemic juvenile idiopathic arthritis reveals distinct biomarkers: a single-center study

Author

Heshuang Qu, Erik Sundberg, Cecilia Aulin, Manoj Neog, Karin Palmblad, Anna Carin Horne, Fredrik Granath, Alexandra Ek, Erik Melén, Mia Olsson, and Helena Erlandsson Harris

Subjects
Systemic juvenile idiopathic arthritis, Cytokines and inflammatory mediators, Inflammation, Proteomics, Ingenuity pathway analysis, High mobility group Box 1, Pediatrics, RJ1-570, Diseases of the musculoskeletal system, RC925-935
Abstract

Abstract Background This study aimed to perform an immunoprofiling of systemic juvenile idiopathic arthritis (sJIA) in order to define biomarkers of clinical use as well as reveal new immune mechanisms. Methods Immunoprofiling of plasma samples from a clinically well-described cohort consisting of 21 sJIA patients as well as 60 age and sex matched healthy controls, was performed by a highly sensitive proteomic immunoassay. Based on the biomarkers being significantly up- or down-regulated in cross-sectional and paired analysis, related canonical pathways and cellular functions were explored by Ingenuity Pathway Analysis (IPA). Results The well-studied sJIA biomarkers, IL6, IL18 and S100A12, were confirmed to be increased during active sJIA as compared to healthy controls. IL18 was the only factor found to be increased during inactive sJIA as compared to healthy controls. Novel factors, including CASP8, CCL23, CD6, CXCL1, CXCL11, CXCL5, EIF4EBP1, KITLG, MMP1, OSM, SIRT2, SULT1A1 and TNFSF11, were found to be differentially expressed in active and/or inactive sJIA and healthy controls. No significant pathway activation could be predicted based on the limited factor input to the IPA. High Mobility Group Box 1 (HMGB1), a damage associated molecular pattern being involved in a series of inflammatory diseases, was determined to be higher in active sJIA than inactive sJIA. Conclusions We could identify a novel set of biomarkers distinguishing active sJIA from inactive sJIA or healthy controls. Our findings enable a better understanding of the immune mechanisms active in sJIA and aid the development of future diagnostic and therapeutic strategies.

Published
2021
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4. Procedures for the content, conduct and format of EULAR/PReS paediatric musculoskeletal ultrasound courses

Author

Maria Antonietta D'Agostino, Esperanza Naredo, Alice Leahy, Hans de Graaf, Rosa Bou, Estibaliz Iglesias, Consuelo Modesto, Karin Palmblad, Giorgia Martini, Séverine Guillaume, Troels Herlin, Dragana Lazarevic, Clara Malattia, Lampros Fotis, Marite Rygg, Denise Pires Marafon, Giovanni Filocamo, Silvia Magni-Manzoni, Sorina Boiu, Nikolay Tzaribachev, Jelena Vojinovic, Lucia Campos, Valentina Muratore, Stefano Lanni, Miroslav Harjacek, Ellen Nordal, Mandica Vidović, Angela Aquilani, Francesca Ardenti Morini, Ellen Dalen Arnstad, Stefan Blazina, Vasiliki Dermentzoglou, Gry Børmark Hoftun, Christian Høst, Emilio Inarejos, Hanan Jadoun, Damjana Ključevšek, Manuela Krumrey, Rolf-Michael Küster, Hartwig Lehmann, Mirea Lopez Corbeto, Anette Lundestad, Marta Mazzoni, Rebecca Nicolai, Soley Omarsdottir, Linda Rossi Semerano, Nina Sande, Betul Sozeri, Giusyda Tarantino, Samar Tharwat, Ralph Trauzeddel, Maria Tsinti, Andrea Uva, Philomine Van Pelt, Marion Van Rossum, Annette von-Scheven-Gete, and Daniel Windschall

Subjects
Medicine
Abstract

Background Despite the worldwide increasing request of education on paediatric musculoskeletal ultrasound (PedMSUS), content, conduct and format of PedMSUS courses have never been internationally agreed.Objectives To produce educational procedures for the conduct, content and format of EULAR/PReS PedMSUS courses.Methods After a systemic literature review and expert opinion collection, a panel of items for the development of procedures on PedMSUS courses was identified. Agreement on the items was assessed through Delphi surveys among a taskforce of 24 members, which included 18 experts in PedMSUS (8 rheumatologists, 1 radiologist, 9 paediatric rheumatologists), 1 methodologist and rheumatologist expert in MSUS, 2 patient research partners, 1 health professional in rheumatology and 2 EMEUNET/EMERGE members, from 8 different European countries. Each item was assessed through a 5-point Likert scale (0, full disagreement; 5, full agreement); agreement was reached for >75% of answers rating 4–5. All items with agreement were included in the preliminary core set of educational procedures, which underwent external assessment by a broader Consensus group (Faculty and Tutors of previous EULAR PedMSUS courses and PReS Imaging Working Party members), through Delphi survey.Results Two Delphi surveys produced the preliminary core set of procedures for basic, intermediate, advanced and teach-the-teachers (TTT) PedMSUS courses. A Delphi survey within the Consensus group produced agreement on the proposed procedures.Conclusions Shared EULAR/PReS procedures for the conduct, content and format of basic, intermediate, advanced and TTT PedMSUS courses were identified on international basis.

Published
2022
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5. Therapeutic administration of etoposide coincides with reduced systemic HMGB1 levels in macrophage activation syndrome

Author

Karin Palmblad, Hanna Schierbeck, Erik Sundberg, Anna-Carin Horne, Helena Erlandsson Harris, Jan-Inge Henter, and Ulf Andersson

Subjects
HMGB1, Macrophage activation syndrome, HLH, FHL, Inflammation, Pathogenesis, Therapeutics. Pharmacology, RM1-950, Biochemistry, QD415-436
Abstract

Abstract Background Macrophage activation syndrome (MAS) is a potentially fatal complication of systemic inflammation. HMGB1 is a nuclear protein released extracellularly during proinflammatory lytic cell death or secreted by activated macrophages, NK cells, and additional cell types during infection or sterile injury. Extracellular HMGB1 orchestrates central events in inflammation as a prototype alarmin. TLR4 and the receptor for advanced glycation end products operate as key HMGB1 receptors to mediate inflammation. Methods Standard ELISA and cytometric bead array-based methods were used to examine the kinetic pattern for systemic release of HMGB1, ferritin, IL-18, IFN-γ, and MCP-1 before and during treatment of four children with critical MAS. Three of the patients with severe underlying systemic rheumatic diseases were treated with biologics including tocilizumab or anakinra when MAS developed. All patients required intensive care therapy due to life-threatening illness. Add-on etoposide therapy was administered due to insufficient clinical response with standard treatment. Etoposide promotes apoptotic rather than proinflammatory lytic cell death, conceivably ameliorating subsequent systemic inflammation. Results This therapeutic intervention brought disease control coinciding with a decline of the increased systemic HMGB1, IFN-γ, IL-18, and ferritin levels whereas MCP-1 levels evolved independently. Conclusion Systemic HMGB1 levels in MAS have not been reported before. Our results suggest that the molecule is not merely a biomarker of inflammation, but most likely also contributes to the pathogenesis of MAS. These observations encourage further studies of HMGB1 antagonists. They also advocate therapeutic etoposide administration in severe MAS and provide a possible biological explanation for its mode of action.

Published
2021
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6. Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part two

Author

Olga Lomakina, Ekaterina Alekseeva, Sania Valieva, Tatiana Bzarova, Irina Nikishina, Elena Zholobova, Svetlana Rodionovskaya, Maria Kaleda, Yasuo Nakagishi, Masaki Shimizu, Mao Mizuta, Akihiro Yachie, Yuko Sugita, Nami Okamoto, Kousuke Shabana, Takuji Murata, Hiroshi Tamai, Eve M. Smith, Peng Yin, Andrea L. Jorgensen, Michael W. Beresford, on behalf of On behalf of the UK JSLE Cohort Study, Antonio Eleuteri, Beatrice Goilav, Laura Lewandowski, Angel Phuti, Dawn Wahezi, Tamar Rubinstein, Caroline Jones, Paul Newland, Stephen Marks, Rachel Corkhill, Diana Ekdawy, Clarissa Pilkington, Kjell Tullus, Chaim Putterman, Chris Scott, Antony C. Fisher, Andrea Jorgensen, Ezgi Deniz Batu, Can Kosukcu, Ekim Taskiran, Sema Akman, Kubra Ozturk, Betul Sozeri, Erbil Unsal, Zelal Ekinci, Yelda Bilginer, Mehmet Alikasifoglu, Seza Ozen, Hanna Lythgoe, Hermine I. Brunner, Gaurav Gulati, Jordan T. Jones, Mekibib Altaye, Jamie Eaton, Mark Difrancesco, Joo Guan Yeo, Jingyao Leong, Loshinidevi D/O Thana Bathi, Thaschawee Arkachaisri, Salvatore Albani, Nagla Abdelrahman, Michael W Beresford, Valentina Leone, UK JSLE study group supported by the National Institute of Health Research Clinical Research Network, Noortje Groot, D. Shaikhani, I. E. M. Bultink, M. Bijl, R. J. E. M. Dolhain, Y. K. O. Teng, E. Zirkzee, K. de Leeuw, R. Fritsch-Stork, S. S. M. Kamphuis, Rachael D. Wright, Reem Abdawani, Laila Al Shaqshi, Ibrahim Al Zakwani, Natali W. Gormezano, David Kern, Oriany L. Pereira, Gladys C. C. Esteves, Adriana M. Sallum, Nadia E. Aikawa, Rosa M. Pereira, Clovis A. Silva, Eloisa Bonfa, Jessica Beckmann, Nora Bartholomä, Nils Venhoff, Philipp Henneke, Ulrich Salzer, Ales Janda, Alina Lucica Boteanu, Sandra Garrote Corral, Alberto Sifuentes Giraldo, Mariluz Gámir Gámir, Antonio Zea Mendoza, Amra Adrovic, Reyhan Dedeoglu, Sezgin Sahin, Kenan Barut, Aida Koka, Funda Oztunc, Ozgur Kasapcopur, Ana Luisa Rodriguez-Lozano, Francisco Rivas-Larrauri, Silvestre García de la Puente, Andressa G. F. Alves, Maria F. D. A. Giacomin, Juliana Farhat, Alfésio L. F. Braga, Adriana M. E. Sallum, Lúcia M. D. A. Campos, Luiz A. A. Pereira, Ana J. D. F. C. Lichtenfels, Clóvis A. Silva, Sylvia C. L. Farhat, Banu Acar, Z. Birsin Ozcakar, Nilgün Çakar, Nermin Uncu, Gökçe Gür, Semanur Özdel, Fatoş Yalçınkaya, Christiaan Scott, Nicky Brice, Peter Nourse, Christine Arango, Angela C. Mosquera, Clara Malagon, Ana P. Sakamoto, Marco F. C. D. Silva, Ananadreia S. Lopes, Gleice C. S. Russo, Adriana E. M. Sallum, Katia Kozu, Eloisa Bonfá, Claudia Saad-Magalhães, Rosa M. R. Pereira, Claudio A. Len, Maria T. Terreri, Deepti Suri, Siyaram Didel, Amit Rawat, Surjit Singh, Despoina Maritsi, MArgarita Onoufriou, Olga Vougiouka, Maria Tsolia, Edi Paleka Bosak, Mandica Vidović, Mirta Lamot, Lovro Lamot, Miroslav Harjaček, Erika Van Nieuwenhove, Adrian Liston, Carine Wouters, Fatemeh Tahghighi, Vahid Ziaee, Seid-Reza Raeeskarami, Francisca Aguiar, Sandra Pereira, Mariana Rodrigues, Cláudia Moura, Gustavo Rocha, Hercília Guimarães, Iva Brito, Rita Fonseca, Gerd Horneff, Ariane Klein, Kirsten Minden, Hans-Iko Huppertz, Frank Weller-Heinemann, Jasmin Kuemmerle-Deschner, J-Peter Haas, Anton Hospach, BIKER collaborative group, Ricardo Menendez-Castro, Boris Huegle, Johannes-Peter Haas, Joost Swart, Gabriella Giancane, Francesca Bovis, Elio Castagnola, Andreas Groll, Daniel J. Lovell, Tom Wolfs, Michael Hofer, Violeta Panaviene, Susan Nielsen, Jordi Anton, Florence Uettwiller, Valda Stanevicha, Maria Trachana, Denise Pires Marafon, Constantin Ailioaie, Elena Tsitsami, Sylvia Kamphuis, Troels Herlin, Pavla Doležalová, Gordana Susic, Berit Flatø, Flavio Sztajnbok, Angela Pistorio, Alberto Martini, Nico Wulffraat, Nicolino Ruperto, Marco Gattorno, Antonio Brucato, Martina Finetti, George Lazaros, Silvia Maestroni, Mara Carraro, Davide Cumetti, Alessandra Carobbio, Monia Lorini, Alessandro Rimini, Renzo Marcolongo, Anna Valenti, Gian Luca Erre, Riccardo Belli, Fiorenzo Gaita, Maria Pia Sormani, Massimo Imazio, Mario Abinun, Nicola Smith, Tim Rapley, Flora McErlane, Lianne Kearsley-Fleet, Kimme L. Hyrich, Helen Foster, Nikolay Tzaribachev, Andrew Zeft, Rolando Cimaz, John Bohnsack, Thomas Griffin, Ruy Carrasco, Jason Dare, Ivan Foeldvari, Richard Vehe, Teresa Simon, Hermine Brunner, S. Verazza, S. Davì, A. Consolaro, A. Insalaco, V. Gerloni, R. Cimaz, F. Zulian, S. Pastore, F. Corona, G. Conti, P. Barone, M. Cattalini, E. Cortis, L. Breda, A. N. Olivieri, A. Civino, R. Podda, D. Rigante, F. La Torre, G. D’Angelo, M. Jorini, R. Gallizzi, M. C. Maggio, R. Consolini, A. De Fanti, M. G. Alpigiani, A. Martini, A. Ravelli, on behalf of Italian Pediatric Rheumatology Study Group, Aysenur Pac Kısaarslan, Zubeyde Gunduz, Ruhan Dusunsel, Ismail Dursun, Hakan Poyrazoglu, Ekaterina Kuchinskaya, Farida Abduragimova, Mikhail Kostik, Erik Sundberg, Soley Omarsdottir, Lena Klevenvall, Helena Erlandsson-Harris, Gokalp Basbozkurt, Ozge Erdemli, Dogan Simsek, Fatih Yazici, Yildirim Karsioglu, Aysen Tezcaner, Dilek Keskin, Huseyin Ozkan, Cengizhan Acikel, Erkan Demirkaya, Ilonka Orbán, Krisztina Sevcic, Valentin Brodszky, Emese Kiss, Ismaiel A. Tekko, Madeleine Rooney, James McElnay, Cliff Taggart, Helen McCarthy, Ryan F. Donnelly, Drug Delivery Group, Mary Slatter, Zohreh Nademi, Mark Friswell, Sharmila Jandial, Terence Flood, Sophie Hambleton, Andrew Gennery, Andrew Cant, Phoi-Ngoc Duong, Isabelle Koné-Paut, Giovanni Filocamo, María Luz Gamir, Helga Sanner, Laura Carenini, Mesut Topdemir, Yildirim Karslioglu, Faysal Gok, Nadezhda Tsurikova, Elena Ligostaeva, Navdha R. Ramchurn, O. Kostareva, I. Nikishina, S. Arsenyeva, S. Rodionovskaya, M. Kaleda, D. Alexeev, Ismail Dursun Dursun, Sara Murias, Estefania Barral, Rosa Alcobendas, Eugenia Enriquez, Agustin Remesal, Jaime de Inocencio, Tania M. Castro, Simone A. Lotufo, Tatjana Freye, Raffaella Carlomagno, Thomas Zumbrunn, Jan Bonhoeffer, Elvira Cannizzaro Schneider, Daniela Kaiser, Michaël Hofer, Véronique Hentgen, Andreas Woerner, Juvenile Inflammatory Rheumatism (JIR) Cohort, Tobias Schwarz, Jens Klotsche, Martina Niewerth, Gerd Ganser, ICON study group, Jerold Jeyaratnam, Nienke ter Haar, Donato Rigante, Fatma Dedeoglu, Ezgi Baris, Sebastiaan Vastert, Joost Frenkel, Jonathan S. Hausmann, Kathleen G. Lomax, Ari Shapiro, Karen L. Durrant, P. A. Brogan, M. Hofer, J. B. Kuemmerle-Deschner, B. Lauwerys, A. Speziale, K. Leon, X. Wei, R. M. Laxer, Sara Signa, Marta Rusmini, Elena Campione, Sabrina Chiesa, Alice Grossi, Alessia Omenetti, Roberta Caorsi, Gianmaria Viglizzo, Isabella Ceccherini, Silvia Federici, Helen Lachmann, Nicola Ruperto, on behalf of PRINTO and Eurofever Registry, Federica Vanoni, on behalf of PRINTO and Eurofever Project, Sonia Melo Gomes, Ebun Omoyinmi, Juan I. Arostegui, Eva Gonzalez-Roca, Despina Eleftheriou, Nigel Klein, Paul Brogan, Stefano Volpi, Elettra Santori, Paolo Picco, Claudia Pastorino, Gillian Rice, Alessandra Tesser, Yanick Crow, Fabio Candotti, Ada B. Sinoplu, Gozde Yucel, Gizem Pamuk, Laura O. Damian, Cecilia Lazea, Mihaela Sparchez, Paulina Vele, Laura Muntean, Adriana Albu, Simona Rednic, Calin Lazar, Leonardo O. Mendonça, Alessandra Pontillo, Jorge Kalil, Fabio M. Castro, Myrthes T. Barros, Manuela Pardeo, Virginia Messia, Fabrizio De Benedetti, Antonella Insalaco, Giorgia Malighetti, Chiara Gorio, Francesca Ricci, Ilaria Parissenti, Paola Montesano, Barbara Bonafini, Veronica Medeghini, Marco Cattalini, Lucio Giordano, Giulia Zani, Rosalba Ferraro, Donatella Vairo, Silvia Giliani, Maria Cristina Maggio, Girolamo Luppino, Giovanni Corsello, Maria Isabel Gonzalez Fernandez, Berta Lopez Montesinos, Adriana Rodriguez Vidal, Juan I. Arostegui Gorospe, Inmaculada Calvo Penades, Nadia K. Rafiq, Karen Wynne, Khalid Hussain, Paul A. Brogan, Elizabeth Ang, Nicholas Ng, Ayla Kacar, Ozge Altug Gucenmez, Balahan Makay, Sevket Erbil Unsal, Yasin Sahin, Tufan Kutlu, Fugen Cullu-Cokugras, Hasret Ayyildiz-Civan, Tulay Erkan, Sana Al Zuhbi, Eiman Abdalla, Ricardo A. Russo, María M. Katsicas, Francesca Minoia, Angelo Ravelli, Sagar Bhattad, Anju Gupta, Vignesh Pandiarajan, Ritambhra Nada, Kaara Tiewsoh, Philip Hawkins, Dorota Rowczenio, Sarka Fingerhutova, Jana Franova, Leona Prochazkova, Eva Hlavackova, Pavla Dolezalova, Havva Evrengül, Selçuk Yüksel, Mustafa Doğan, Dolunay Gürses, Harun Evrengül, Silvia De Pauli, Serena Pastore, Anna Monica Bianco, Giovanni Maria Severini, Andrea Taddio, Alberto Tommasini, Svetlana O. Salugina, Evgeny Fedorov, Elena Kamenets, Ekaterina Zaharova, Tatiana Sleptsova, Ekaterina Alexeeva, Kirill Savostyanov, Alexander Pushkov, Tatyana Bzarova, Saniya Valieva, Rina Denisova, Kseniya Isayeva, Evgeniya Chistyakova, Margarita Soloshenko, Elena Kaschenko, Utako Kaneko, Chihaya Imai, Akihiko Saitoh, Vitor A. Teixeira, Filipa O. Ramos, Manuela Costa, Yonatan Butbul Aviel, Shafe Fahoum, Riva Brik, Zeynep Birsin Özçakar, Banu Acar Celikel, Fatos Yalcinkaya, Benedetta Schiappapietra, Sergio Davi’, Federica Mongini, Luisa Giannone, Cecilia Bava, Maria Giannina Alpigiani, Alessandro Consolaro, Dragana S. Lazarevic, Jelena Vojinovic, Jelena Basic, Valentina Muratore, Valentina Marzetti, Neus Quilis, Belen Serrano Benavente, Alessandra Alongi, Adele Civino, Lorenzo Quartulli, Giedre Januskeviciute, Pieter van Dijkhuizen, N. Groot, W. van Dijk, A. Kardolus, Raul Gutiérrez Suárez, Ellen B. Nordal, Veronika G. Rypdal, Lillemor Berntson, Maria Ekelund, Kristiina Aalto, Suvi Peltoniemi, Marek Zak, Mia Glerup, Ellen D. Arnstad, Anders Fasth, Marite Rygg, the Nordic Study Group of Pediatric Rheumatology (NoSPeR), Ana Catarina Duarte, Sandra Sousa, Lídia Teixeira, Ana Cordeiro, Mª José Santos, Ana Filipa Mourão, Maria José Santos, Mónica Eusébio, Ana Lopes, Filipa Oliveira-Ramos, Manuel Salgado, Paula Estanqueiro, José Melo-Gomes, Fernando Martins, José Costa, Carolina Furtado, Ricardo Figueira, Jaime C. Branco, João E. Fonseca, Helena Canhão, Ana F. Mourão, Maria Jose Santos, Andrea Coda, Samuel Cassidy, Kerry West, Gordon Hendry, Debra Grech, Julie Jones, Fiona Hawke, Davinder Singh Grewal, Charlene Foley, Orla Killeen, Emma MacDermott, Douglas Veale, Ursula Fearon, Dilek Konukbay, Ela Tarakci, Nilay Arman, Sezgin Şahin, Jane Munro, Esi Morgan, Meredith Riebschleger, Jennifer Horonjeff, Vibeke Strand, Clifton Bingham, Ma. Theresa M. Collante, Margarita Ganeva, Stefan Stefanov, Albena Telcharova, Dimitrina Mihaylova, Radoslava Saraeva, Reni Tzveova, Radka Kaneva, Adelina Tsakova, Katya Temelkova, GRANT Medical University, Sofia 68/, Maria Mercedes C. Picarelli, Luiz C. Danzmann, Florencia Barbé-Tuana, Lucas K. Grun, Marcus H. Jones, Marijan Frković, Karla Ištuk, Ika Birkić, Saša Sršen, Marija Jelušić, Alan Easton, Rachael Quarmby, Raju Khubchandani, Mercedes Chan, Radoslav Srp, Katerina Kobrova, Dana Nemcova, Jozef Hoza, Michal Uher, Melania Saifridova, Lenka Linkova, Sirirat Charuvanij, Isree Leelayuwattanakul, Thita Pacharapakornpong, Sakda A.-O. Vallipakorn, Butsabong Lerkvaleekul, Soamarat Vilaiyuk, Stefano Lanni, Sergio Davì, Randy Q. Cron, Chiara Passarelli, Elisa Pisaneschi, Antonio Novelli, Claudia Bracaglia, Ivan Caiello, Kathy de Graaf, Florence Guilhot, Walter Ferlin, Grant Schulert, Alexi A. Grom, Robert Nelson, Cristina de Min, Dirk Holzinger, Christoph Kessel, Ndate Fall, Alexei Grom, Wilco de Jager, Raffaele Strippoli, Anna Horne, Stephan Ehl, Sandra Ammann, Kai Lehmberg, Karin Beutel, Dirk Foell, AnnaCarin Horne, Laura Pagani, Graciela Espada, Yi-jin Gao, Susan Shenoi, Sheila Weitzman, Giusi Prencipe, Antonia Pascarella, Walter G. Ferlin, Laurence Chatel, Philippe Jacqmin, Kathy De Graaf, Maria Ballabio, Zoë Johnson, Geneviève Lapeyre, Fabrizio de Benedetti, de Min Cristina, Hiroyuki Wakiguchi, Shunji Hasegawa, Reiji Hirano, Fumiko Okazaki, Tamaki Nakamura, Hidenobu Kaneyasu, Shouichi Ohga, Kazuko Yamazaki, Tomo Nozawa, Taichi Kanetaka, Shuichi Ito, Shumpei Yokota, Kirsty McLellan, Ishbel MacGregor, Neil Martin, Joyce Davidson, Sandra Hansmann, Andreas Eikelberg, Iris Haug, Sabrina Schuller, Susanne M. Benseler, Single Hub and Access point for paediatric Rheumatology in Europe (SHARE), Liliia S. Nazarova, Kseniia V. Danilko, Viktor A. Malievsky, Tatiana V. Viktorova, Angela Mauro, Angela Barnicoat, Jane Hurst, Nathalie Canham, Sandrine Lacassagne, Anastasia Wiener, Boris Hügle, Bernd Denecke, Ivan Costa-Filho, Johannes Peter Haas, Klaus Tenbrock, David Popp, Arjan Boltjes, Frank Rühle, Stefanie Herresthal, Femke van Wijk, Joachim Schultze, Monika Stoll, Luisa Klotz, Thomas Vogl, Johannes Roth, Estefania Quesada-Masachs, Daniel Álvarez de la Sierra, Marina Garcia Prat, Ana M. Marín Sánchez, Ricardo Pujol Borrell, Sara Marsal Barril, Mónica Martínez Gallo, Consuelo Modesto Caballero, Iryna Chyzheuskaya, Lyudmyla M. Byelyaeva, Rostislav M. Filonovich, Helena K. Khrustaleva, Larisa I. Zajtseva, Tamara M. Yuraga, Thomas Giner, Lukas Hackl, Julia Albrecht, Reinhard Würzner, Juergen Brunner, Marta Minute, Fulvio Parentin, Agostino Nocerino, Mette Nørgaard, Mikel Alberdi-Saugstrup, Marek S. Zak, Susan M. Nielsen, Ellen Nordal, Klaus G. Müller, Nordic Study Group of Pediatric Rheumatology (NoSPeR), Mojca Zajc Avramovič, Vita Dolžan, Nataša Toplak, Tadej Avčin, N. Ruperto, D. J. Lovell, C. Wallace, M. Toth, I. Foeldvari, J. Bohnsack, D. Milojevic, C. Rabinovich, D. Kingsbury, K. Marzan, P. Quartier, K. Minden, E. Chalom, G. Horneff, R. M. Kuester, J. Dare, M. Heinrich, H. Kupper, J. Kalabic, H. I. Brunner, on behalf of PRINTO and PRCSG, Ruben Burgos-Vargas, Tamas Constantin, Joke Dehoorne, Valda Stanevica, Katarzyna Kobusinska, Zbigniew Zuber, Richard Mouy, Ingrida Rumba-Rozenfelde, Chantal Job-Deslandre, Ronald Pederson, Jack Bukowski, Tina Hinnershitz, Bonnie Vlahos, Paula Keskitalo, Salla Kangas, Paula Vähäsalo, Raul A. Chavez Valencia, David Martino, Anne-Louise Ponsonby, Rachel Chiaroni-Clarke, Braydon Meyer, Roger C. Allen, Jonathan D. Akikusa, Jeffrey M. Craig, Richard Saffrey, Justine A. Ellis, Carol Wallace, Yosef Uziel, Gary Sterba, Rayfel Schneider, Ricardo Russo, Athimalaipet V. Ramanan, Jana Pachlopnik Schmid, Kim E Nichols, Paivi Miettunen, Toshiyuki Kitoh, Norman T. Ilowite, Jan-Inge Henter, Alexei A Grom, Edward M. Behrens, Tadej Avcin, Maurizio Aricò, Sriharsha Grevich, Peggy Lee, Sarah Ringold, Brian Leroux, Hannah Leahey, Megan Yuasa, Jessica Foster, Jeremy Sokolove, Lauren Lahey, William Robinson, Joshua Newson, Anne Stevens, Stephanie J. W. Shoop, Suzanne M. M. Verstappen, Wendy Thomson, Janet E. McDonagh, CAPS, Timothy Beukelman, Yuki Kimura, Marc Natter, Norm Ilowite, Kelly Mieszkalski, Grendel Burrell, Brian Best, Helen Bristow, Shannon Carr, Anne Dennos, Rachel Kaufmann, Laura Schanberg, for the CARRA Registry Investigators, Gabriele Simonini, Francesca Lancini, Margaux Gerbaux, Phu-Quoc Lê, Laurence Goffin, Valérie Badot, Céline La, Laure Caspers, François Willermain, Alina Ferster, Maria Ceci, Francesco Licciardi, Marco Turco, Francesca Santarelli, Davide Montin, Claudia Toppino, Clotilde Alizzi, Bruno Papia, Beatrice Vergara, Umberto Corpora, Luca Messina, Maria Tsinti, Vasiliko Dermentzoglou, Panagiotis Tziavas, Marija Perica, Lana Tambić Bukovac, Mustafa Çakan, Nuray Aktay Ayaz, Gonca Keskindemirci, Michael Lang, Catherine Laing, Susanne Benseler, Tommy Gerschman, Nadia Luca, Heinrike Schmeling, Anastasia Dropol, Jaymi Taiani, Nicole Johnson, Brian Rusted, Panagiota Nalbanti, Polyxeni Pratsidou, Grigoris Pardalos, Vasiliki Tzimouli, Anna Taparkou, Maria Stavrakidou, Fotios Papachristou, Florence Kanakoudi-Tsakalidou, Peter Bale, Emily Robinson, Jason Palman, Elizabeth Ralph, Kimberly Gilmour, Clare Heard, Lucy R. Wedderburn, Yara Barrense-Dias, Antonarakis Gregory, Dhouib Amira, Scolozzi Paolo, Hanquinet Sylviane, Hofer Michaël, Nataliya Panko, Salah Shokry, Liudmila Rakovska, Sally Pino, Adriana Diaz-Maldonado, Pilar Guarnizo, Sofia Torreggiani, Paolo Cressoni, Umberto Garagiola, Giancarla Di Landro, Giampietro Farronato, Fabrizia Corona, Samantha Bell, Parveen Bhatti, Lee Nelson, Beth A. Mueller, T. A. Simon, A. Baheti, N. Ray, Z. Guo, Anasuya Hazra, Thomas Stock, Ronnie Wang, Charles Mebus, Christine Alvey, Manisha Lamba, Sriram Krishnaswami, Umberto Conte, Min Wang, Daniel Kingsbury, Elena Koskova, Elzbieta Smolewska, Richard K. Vehe, Daniel Lovell, Tomohiro Kubota, Junko Yasumura, Toshitaka Kizawa, Masato Yashiro, Tsuyoshi Yamatou, Yuichi Yamasaki, Syuji Takei, Yoshifumi Kawano, Ulrika Järpemo Nykvist, Bo Magnusson, Rikard Wicksell, Karin Palmblad, Gunnar L. Olsson, Mohammadreza Modaressi, Mohammad-Hassan Moradinejad, Valentina Seraya, Alisa Vitebskaya, Veronica Moshe, Gil Amarilyo, Liora Harel, Phillip J Hashkes, Amir Mendelson, Noa Rabinowicz, Yonit Reis, Zane Dāvidsone, Arina Lazareva, Ruta Šantere, Dace Bērziņa, Valda Staņēviča, Giulia Camilla Varnier, Susan Maillard, Cristina Ferrari, Silvia Zaffarano, Juvenile Dermatomyositis Research Group and European Federation of Immunological Societies, Judith Wienke, Felicitas Bellutti Enders, Lucas L. van den Hoogen, Jorre S. Mertens, Timothy R. Radstake, Henny G. Hotten, Ruth Fritsch, Lucy Wedderburn, Kiran Nistala, Berent Prakken, Annet van Royen-Kerkhof, Mohammad Alhemairi, Mohammed Muzaffer, Pieter Van Dijkhuizen, Claire T. Deakin, Stefania Simou, Maria De Iorio, Qiong Wu, Tania Amin, Lee Dossetter, Juvenile Dermatomyositis Research Group (JDRG), Raquel Campanilho-Marques, Claire Deakin, Clarissa A. Pilkington, on behalf of Juvenile Dermatomyositis Research Group (JDRG), Silvia Rosina, Sirisucha Soponkanaporn, on behalf of the UK Juvenile Dermatomyositis Research Group (JDRG), Zehra S. Arıcı, Gökçen D. Tuğcu, Ezgi D. Batu, Hafize E. Sönmez, Deniz Doğru-Ersöz, Beril Talim, Nural Kiper, Seza Özen, Alexander Solyom, Ezgi Batu, John Mitchell, Ariana Kariminejad, Fatemeh Hadipour, Zahra Hadipour, Marta Torcoletti, Carlo Agostoni, Maja Di Rocco, Pranoot Tanpaiboon, Andrea Superti-Furga, Luisa Bonafé, Nur Arslan, Norberto Guelbert, Karoline Ehlert, Giedre Grigelioniene, Ratna Puri, Edward Schuchman, Pilar Gomez, Tatiana Gonzalez, Ricardo Yepez, Camilo Vargas, GRIP study group, Falcini Fernanda, Gemma Lepri, Alessandra Ferrari, Marco Matucci-Cerinic, Antonella Meini, Gian Marco Moneta, Emiliano Marasco, Rebecca Nicolai, Luisa Bracci-Laudiero, Olga Kopchak, Alexander Mushkin, Alexey Maletin, Catalina Mosquera, Rita A. Amorim, Juliana Molina, Gustavo Moreira, Flávia H. Santos, Melissa Fraga, Livia Keppeke, Vanessa M. Silva, Camila Hirotsu, Sergio Tufik, Maria Teresa Terreri, Vinícius L. Braga, Maria Beatriz Fonseca, Vania Schinzel, Maria Teresa R. Terreri, Liliana Jorge, Liana Guerra, Edson Amaro Junior, Maria Cristina Castiglione, Alessandra Tricarico, Emily Boulter, Andre Schultz, Kevin Murray, Fernanda Falcini, Stefano Stagi, Eleonora Bellucci, Ingrid H. R. Grein, Gecilmara Pileggi, Natália B. F. Pinto, Aline L. de Oliveira, Lyudmila Belyaeva, Rostislav Filonovich, Helena Khrustaleva, Larisa Zajtseva, Jaanika Ilisson, Chris Pruunsild, Olivier Gilliaux, Francis Corazza, Christophe Lelubre, on behalf of PANLAR Pediatric Rheumatology Study Group, Zoilo Morel, Claudia Saad-Magalhães C, Luis Lira, Mabel Ladino, Ruth Eraso, Ivonne Arroyo, Clovis Silva, Carlos Rose, and PANLAR Pediatric Rheumatology Study Group

Subjects
Pediatrics, RJ1-570, Diseases of the musculoskeletal system, RC925-935
Published
2017
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7. Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part three

Author

Riccardo Papa, Alessandro Consolaro, Francesca Minoia, Roberta Caorsi, Gianmichele Magnano, Marco Gattorno, Angelo Ravelli, Paolo Picco, Roberto Pillon, Denise Pires Marafon, Lidia Meli, Claudia Bracaglia, Andrea Taddio, Fabrizio De Benedetti, Enes Turan, Sara Sebnem Kilic, Yasuhiko Itoh, Tomoko Shigemori, Shingo Yamanishi, Hidehiko Nagasaki, Ela Tarakci, Nilay Arman, Devrim Tarakci, Yusuf S. Akgul, Ozgur Kasapcopur, Emily Wilson, Hanna Lythgoe, Eve Smith, Jenny Preston, Michael W. Beresford, Lynn R. Spiegel, Jennifer Stinson, Mark Connelly, Adam Huber, Nadia Luca, Argerie Tsimicalis, Stephanie Luca, Naweed Tajuddin, Roberta Berard, Julie Barsalou, Sarah Campillo, Brian Feldman, Shirley Tse, Paul Dancey, Ciaran Duffy, Nicole Johnson, Patrick McGrath, Natalie Shiff, Lori Tucker, Charles Victor, Chitra Lalloo, Lauren Harris, Joseph Cafazzo, Kristin Houghton, Ronald Laxer, Madeleine Rooney, Roisin Campbell, Catherine Wright, Wineke Armbrust, Otto Lelieveld, Jolanda Tuinstra, Nico Wulffraat, Joyce Bos, Jeanette Cappon, Marion van Rossum, Mariët Hagedoorn, Anna Vermé, Ylva Lampela, Ayse Huri Ozdogan, S. Ugurlu, K. Barut, A. Androvic, O. Kasapçopu, Jody Etheridge, Katie Dobson, Sue Kemp, AnnaCarin Horne, Karin Palmblad, Malin Höglund, Natalia Stepanenko, Svetlana Salugina, Evgeny Fedorov, Irina Nikishina, Maria Kaleda, Kenan Barut, Amra Adrovic, Sezgin Sahin, Laurence Toumoulin, Johnny Frossard, Stephanie Archimbaut, Anne Paitier, Rolande Guastalli, Severine Guillaume Czitrom, Sirirat Charuvanij, Chollada Chaiyadech, Takako Miyamae, Hisashi Yamanaka, Cecile Picard, Guillaume Thouvenin, Caroline Kannengiesser, Jean-Christophe Dubus, Nadia Jeremiah, Frédéric Rieux-Laucat, Bruno Crestani, Véronique Secq, Christelle Ménard, Martine Reynaud-Gaubert, Françoise Thivolet-Bejui, Philippe Reix, Alexandre Belot, Ezgi Deniz Batu, Hafize Emine Sonmez, Abdulsamet Erden, Ekim Z. Taskiran, Omer Karadag, Umut Kalyoncu, İbrahim Oncel, Berkan Kaplan, Zehra Serap Arici, Cagri Mesut Temucin, Haluk Topaloglu, Yelda Bilginer, Mehmet Alikasifoglu, Seza Ozen, Lien Van Eyck, Ellen De Langhe, Isabelle Jéru, Erika Van Nieuwenhove, Vasiliki Lagou, Paul J. Baker, Jocelyn Garcia-Perez, James Dooley, Lien De Somer, Raf Sciot, Pierre-Yves Jeandel, Julia Ruuth-Praz, Bruno Copin, Myrna Medley-Hashim, Andre Megarbane, Sinisa Savic, An Goris, Serge Amselem, Adrian Liston, Seth Masters, Carine Wouters, Nami Okamoto, Yuko Sugita, Kousuke Shabana, Takuji Murata, Hiroshi Tamai, Juliana Ferenczová, Erika Banóova, Pavol Mrážik, Veronika Vargova, Dubravko Bajramovic, Ksenija Stekic Novacki, Kristina Potocki, Marijan Frkovic, Marija Jelusic, Olga Kostareva, Svetlana Arsenyeva, Anna Shapovalenko, Lennart Jans, Nele Herregods, Jacob Jaremko, Rik Joos, Joke Dehoorne, Xenofon Baraliakos, Sofia Ramiro, Julio C. Casasola-Vargas, Désirée van der Heijde, Robert Landewé, Ruben Burgos-Vargas, Shirley M. Tse, Gerd Horneff, Kristina Unnebrink, Jaclyn K. Anderson, Aysenur Paç Kisaarslan, Betül Sözeri, Zübeyde Gündüz, Gökmen Zararsız, Hakan Poyrazoğlu, Ruhan Düşünsel, Kazutaka Ouchi, Shinji Akioka, Hiroshi Kubo, Norio Nakagawa, Hajime Hosoi, Lovro Lamot, Fran Borovecki, Sanja Kapitanovic, Kristina Gotovac, Mandica Vidovic, Mirta Lamot, Edi Paleka Bosak, Miroslav Harjacek, Ricardo A. Russo, María M. Katsicas, Ruben Burgos Vargas, Ana L. Ortiz-Peyegahud, Zhang Pingping, Mou Yikun, Qi Jun, Jiang Yutong, Gu Jieruo, Mikhail M. Kostik, Shilova Ekaterina, Ilia Avrusin, Yuriy Korin, Olga Kopchak, Eugenia Isupova, Irina Chikova, Panova Tatyana, Margarita Dubko, Vera Masalova, Ludmila Snegireva, Tatyana Kornishina, Olga Kalashnikova, Vyacheslav Chasnyk, Tatyana Likhacheva, N. Ruperto, H. I. Brunner, P. Quartier, T. Constantin, E. Alexeeva, R. Schneider, I. Kone-Paut, K. Schikler, K. Marzan, N. Wulffraat, S. Padeh, V. Chasnyk, C. Wouters, J. B. Kuemmerle-Deschner, T. Kallinich, B. Lauwerys, E. Haddad, E. Nasonov, M. Trachana, O. Vougiouka, K. Leon, A. Speziale, K. Lheritier, E. Vritzali, A. Martini, D. Lovell, PRINTO/PRCSG, Nienke Ter Haar, Rianne Scholman, Wilco de Jager, Tamar Tak, Pieter Leliefeld, Bas Vastert, Sytze de Roock, Ariane de Ganck, Nadia Ryter, Miha Lavric, Dirk Foell, Renee F. Modica, Kathleen G. Lomax, Pamela Batzel, Armelle Cassanas, Melissa E. Elder, Rina Denisova, Ekaterina Alexeeva, Saniya Valieva, Tatyana Bzarova, Kseniya Isayeva, Tatyana Sleptsova, Olga Lomakina, Alexandra Chomahidze, Margarita Soloshenko, Meyry Shingarova, Elena Kachshenko, Wilco De Jager, Sebastiaan J. Vastert, Gerdien Mijnheer, Berent J. Prakken, Nico M. Wulffraat, Hafize E. Sönmez, Asuman N. Karhan, Ezgi D. Batu, Zehra S. Arıcı, Ersin Gümüş, Hülya Demir, Aysel Yüce, Seza Özen, Jasmina Ahluwalia, Bhavneet Bharti, Sweta Rajpal, Varun Uppal, Alaknanda Walia, Surjit S. Samlok, Narender Kumar, Clarissa C. Valões, Beatriz C. Molinari, Ana Claudia G. Pitta, Natali W. Gormezano, Sylvia C. Farhat, Kátia Kozu, Adriana M. Sallum, Simone Appenzeller, Ana Paula Sakamoto, Maria T. Terreri, Rosa M. Pereira, Claudia S. Magalhães, Cássia Maria Barbosa, Francisco Hugo Gomes, Eloisa Bonfá, Clovis A. Silva, Kubra Ozturk, Zelal Ekinci, Maie Helal, Natalia Cabrera, Jean Christophe Lega, Jocelyne Drai, Rene Ecochard, O. V. Shpitonkova, N. S. Podchernyaeva, Y. O. Kostina, N. G. Dashkova, M. K. Osminina, Gozde Yucel, Ahmet Arvas, Nandini Moorthy, Paraskevi Dimou, Angela Midgley, Matthew Peak, Simon C. Satchell, Rachael D. Wright, Rachel Corkhill, Eve M. Smith, Sagar Bhattad, Amit Rawat, Surjit Singh, Anju Gupta, Deepti Suri, Martin de Boer, Taco Kuijpers, Vignesh Pandiarajan, Sapna Sandal, Sebastian Giraldo, Roy Sanguino, Adriana S. Diaz, Selcuk Uzuner, Gizem Durcan, Ali Guven Kilicoglu, Ayhan Bilgic, Kayhan Bahali, Sinem Durmus, Hafize Uzun, Nur Canpolat, Salim Caliskan, Lale Sever, Tomomi Sato, Fuminori Kimura, Wafaa Suwairi, Reem Abdwani, Abdulaziz Al Rowais, Jubran Al qanatish, Abdulrahman Al Asiri, Ekaterina Gaidar, Mikhail Kostik, Elena Serogodskaya, Tatyana Nikitina, Evgenia Isupova, Elham Sardar, Perrine Dusser, Antoine Rousseau, Marc Labetoulle, Emanuel Barreau, Bahram Bodaghi, Isabelle Kone-Paut, Ivan Foeldvari, Jordi Anton, Rosa Bou, Sheila Angeles-Han, Regitze Bangsgaard, Gabriele Brumm, Tamas Constantin, Clive Edelsten, Jens Klotsche, Kirsten Minden, Elisabetta Miserocchi, Susan Nielsen, Gabriele Simonini, Arnd Heiligenhaus, Juan Manuel Mosquera Angarita, Carmen Garcia de Vicuña, Maria Victoria Hernandez, Alfredo Adan, Victor Llorens, Rosa Alcobendas, Susana Noval, Juan Carlos Lopez Robledillo, Isabel Valls, Mari Carmen Pinedo, Alejandro Fonollosa, Jaime de Inocencio, Pilar Tejada, Beatriz Bravo, Manuel Torribio, María Jesús García de Yebenes, Jordi Antón, Uveitis Working Group of the Spanish Pediatric Rheumatology Society, Lorenza Maria Argolini, Irene Pontikaki, Maria Orietta Borghi, Laura Cesana, Barbara Castiglioni, Maurizio Gattinara, Pierluigi Meroni, Pierre Quartier, Veronique Despert, Sylvaine Poignant, Amandine Baptiste, Caroline Elie, Laurent Kodjikian, Dominique Monnet, Michel Weber, Laura Moal, LuuLy Pham, Emmanuel Barreau, Cherif Titah, Pascal Dureau, Vanessa Cecchin, Maria Elisabetta Zannin, Daniele Ferrari, Francesco Comacchio, Rolando Cimaz, Fernanda Falcini, Antonella Petaccia, Stefania Viola, Luciana Breda, Francesco La Torre, Fabio Vittadello, Giorgia Martini, Francesco Zulian, Caroline Galeotti, Guillaume Sarrabay, Olivier Fogel, Isabelle Touitou, Corinne Miceli-Richard, Isabelle Koné-Paut, Hala Etayari, Hashad Soad, Ihab El Kadry, Habibullah Eatamadi, Kais AlAlgawi, Mustafa Al Maini, Khulood Khawaja, Sophie Van den Berghe, Ilse de Schryver, Ann Raes, Lídia L. C. Teixeira, Ana Duarte, Sandra Sousa, Filipe Vinagre, Maria J. Santos, Nataly S. Shevchenko, Ludmila F. Bogmat, Marina V. Demyanenko, Navdha R. Ramchurn, Mark Friswell, Rebecca A. James, Lucy R. Wedderburn, Reshma Pattani, Clarissa A. Pilkington, Sandrine Compeyrot-Lacassagne, Ana V. Villarreal, Nydia Acevedo, Enrique Faugier, Rocio Maldonado, Dilek Yılmaz, Hilal Bektaş Uysal, Elena Kamenets, Ekaterina Zaharova, Stefka Radenska-Lopovok, Joao Nascimento, Helena Sofia, Carla Zilhão, Rui Almeida, Margarida Guedes, Murat Deveci, Svetlana Rodionovskaya, Vera Vinnikova, Irina Tsymbal, Edyta Olesińska, Jacek Postępski, Agnieszka Mroczkowska-Juchkiewicz, Agnieszka Pawłowska-Kamieniak, Beata Chrapko, Damjana Ključevšek, Nina Emeršič, Nataša Toplak, Tadej Avčin, Faina Rokhlina, Galina Glazyrina, Natalia Kolyadina, Kwangnam Kim, Sinae Eom, Daeyoung Kim, Jungwoo Rhim, Francesca Ricci, Paola Montesano, Barbara Bonafini, Veronica Medeghini, Ilaria Parissenti, Antonella Meini, Marco Cattalini, Paolo Airò, Nataliya Panko, Nataliya Shevchenko, Iryna Lebec, Yevgeniya Zajceva, Sara Rostlund, Marie André, Takuma Hara, Takayuki Kishi, Yumi Tani, Aki Hanaya, Satoru Nagata, Velma Selmanovic, Aida Omercahic-Dizdarevic, Adisa Cengic, Almira Cosickic, Aida Omerčahić Dizdarević, Gemma Lepri, Clara Malattia, Eleonora Bellucci, Marco Matucci-Cerinic, Anton Solovyev, Elena Fedotova, Ana Victoria Villarreal, Talia Diaz, Yuridiana Ramirez, Teresa Giani, Achille Marino, Daniel Hunt, Muthana Al Obaidi, Veli Veli, Charalampia Papadopoulou, Jochen Kammermeier, Anna Poluha, Gangadhara C. Bharmappanavara, Alison Kelly, Lindsay Shaw, Giovanna Ferrara, Michele Luzzati, Mattia Giovannini, Liliana Jurado, Juliana Chamorro, Lorena Sarmiento, Ester Conversano, Maria Francesca Gicchino, Giulia Macchini, Carmela Granato, Assunta Tirelli, Alma N. Olivieri, Marija Perica, Lana Tambić Bukovac, Reza Sinaei, Vadood Javadi Parvaneh, Reza Shiari, Khosro Rahmani, Fatemeh F. Mehregan, Mehrnoush Hassas Yeganeh, Inmaculada Calvo Penadés, Berta López Montesinos, Ma Isabel González Fernández, Adriana Rodríguez Vidal, Anand Prahalad Rao, Ayesha Romana, Jyothi Raghuram, Ankur Kumar, Vishali Gupta, Elif Comak, Gülşah Kaya Aksoy, Aygen Yılmaz, Atike Atalay, Mustafa Koyun, Reha Artan, Sema Akman, Maria I. Kaleda, Irina P. Nikishina, Sergei K. Soloviev, Victor A. Malievsky, Ekaterina V. Nikolaeva, Agnieszka Gazda, Beata Kołodziejczyk, Lidia Rutkowska-Sak, Angela Mauro, Pierluigi Marzuillo, Stefano Guarino, and Angela La Manna

Subjects
Pediatrics, RJ1-570, Diseases of the musculoskeletal system, RC925-935
Published
2017
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8. Retraction Note to: High systemic levels of the cytokine-inducing HMGB1 isoform secreted in severe macrophage activation syndrome

Author

Karin Palmblad, Hanna Schierbeck, Erik Sundberg, Anna-Carin Horne, Helena Erlandsson Harris, Jan-Inge Henter, Daniel J. Antoine, and Ulf Andersson

Subjects
Therapeutics. Pharmacology, RM1-950, Biochemistry, QD415-436
Abstract

This article has been retracted. Please see the Retraction Notice for more detail: https://doi.org/10.1186/s10020-020-00263-2

Published
2020
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10. Therapeutic administration of etoposide coincides with reduced systemic HMGB1 levels in macrophage activation syndrome

Author

Helena Erlandsson Harris, Karin Palmblad, Erik Sundberg, AnnaCarin Horne, Hanna Schierbeck, Ulf Andersson, and Jan-Inge Henter

Subjects
Male, 0301 basic medicine, Pathogenesis, Systemic inflammation, Biochemistry, chemistry.chemical_compound, 0302 clinical medicine, HMGB1 Protein, Child, Genetics (clinical), Etoposide, HMGB1, biology, Treatment Outcome, Macrophage activation syndrome, Child, Preschool, 030220 oncology & carcinogenesis, Cytokines, Molecular Medicine, Female, Inflammation Mediators, medicine.symptom, Immunosuppressive Agents, Research Article, medicine.drug, HLH, Adolescent, FHL, chemical and pharmacologic phenomena, Inflammation, RM1-950, QD415-436, Proinflammatory cytokine, 03 medical and health sciences, Tocilizumab, Intensive care, Genetics, medicine, Humans, Molecular Biology, business.industry, medicine.disease, Antineoplastic Agents, Phytogenic, 030104 developmental biology, chemistry, Immunology, biology.protein, Therapeutics. Pharmacology, business, Biomarkers
Abstract

Background Macrophage activation syndrome (MAS) is a potentially fatal complication of systemic inflammation. HMGB1 is a nuclear protein released extracellularly during proinflammatory lytic cell death or secreted by activated macrophages, NK cells, and additional cell types during infection or sterile injury. Extracellular HMGB1 orchestrates central events in inflammation as a prototype alarmin. TLR4 and the receptor for advanced glycation end products operate as key HMGB1 receptors to mediate inflammation. Methods Standard ELISA and cytometric bead array-based methods were used to examine the kinetic pattern for systemic release of HMGB1, ferritin, IL-18, IFN-γ, and MCP-1 before and during treatment of four children with critical MAS. Three of the patients with severe underlying systemic rheumatic diseases were treated with biologics including tocilizumab or anakinra when MAS developed. All patients required intensive care therapy due to life-threatening illness. Add-on etoposide therapy was administered due to insufficient clinical response with standard treatment. Etoposide promotes apoptotic rather than proinflammatory lytic cell death, conceivably ameliorating subsequent systemic inflammation. Results This therapeutic intervention brought disease control coinciding with a decline of the increased systemic HMGB1, IFN-γ, IL-18, and ferritin levels whereas MCP-1 levels evolved independently. Conclusion Systemic HMGB1 levels in MAS have not been reported before. Our results suggest that the molecule is not merely a biomarker of inflammation, but most likely also contributes to the pathogenesis of MAS. These observations encourage further studies of HMGB1 antagonists. They also advocate therapeutic etoposide administration in severe MAS and provide a possible biological explanation for its mode of action.

Published
2021

11. Tapering Canakinumab Monotherapy in Patients With Systemic Juvenile Idiopathic Arthritis in Clinical Remission: Results From a Phase IIIb/IV Open‐Label, Randomized Study

Author

Karin Palmblad, Gerd Horneff, Ken Abrams, X Wei, Rayfel Schneider, Vyacheslav Chasnyk, Alberto Martini, Jordi Anton, Katherine Marzan, Carine Wouters, Tamás Constantin, Ekaterina Alexeeva, Hermine I. Brunner, Pierre Quartier, Nicolino Ruperto, Alan Slade, and Nico M Wulffraat

Subjects
Male, medicine.medical_specialty, Adolescent, Immunology, Full Length, Arthritis, Antibodies, Monoclonal, Humanized, law.invention, 03 medical and health sciences, 0302 clinical medicine, Deprescriptions, Rheumatology, Randomized controlled trial, law, Internal medicine, medicine, Immunology and Allergy, Humans, In patient, 030212 general & internal medicine, Child, Glucocorticoids, 030203 arthritis & rheumatology, Drug Tapering, business.industry, Remission Induction, medicine.disease, Pediatric Rheumatology, Pediatric Rheumatology International Trials Organization (PRINTO), the Pediatric Rheumatology Collaborative Study Group (PRCSG), Arthritis, Juvenile, Discontinuation, Canakinumab, Methotrexate, Antirheumatic Agents, Child, Preschool, Female, Open label, Inactive disease, business, medicine.drug
Abstract

OBJECTIVE: To evaluate the efficacy and safety of 2 canakinumab monotherapy tapering regimens in order to maintain complete clinical remission in children with systemic juvenile idiopathic arthritis (JIA). METHODS: The study was designed as a 2-part phase IIIb/IV open-label, randomized trial. In the first part, patients received 4 mg/kg of canakinumab subcutaneously every 4 weeks and discontinued glucocorticoids and/or methotrexate as appropriate. Patients in whom clinical remission was achieved (inactive disease for at least 24 weeks) with canakinumab monotherapy were entered into the second part of the trial, in which they were randomized 1:1 into 1 of 2 treatment arms. In arm 1, the dose of canakinumab was reduced from 4 mg/kg to 2 mg/kg and then to 1 mg/kg, followed by discontinuation. In arm 2, the 4 mg/kg dose interval was prolonged from every 4 weeks, to every 8 weeks, and then to every 12 weeks, followed by discontinuation. In both arms, canakinumab exposure could be reduced provided systemic JIA remained in clinical remission for 24 weeks with each step. The primary objective was to assess whether >40% of randomized patients in either arm maintained clinical remission of systemic JIA for 24 weeks in the first part of the study. RESULTS: In part 1 of the study, 182 patients were enrolled, with 75 of those patients randomized before entering part 2 of the trial. Among the 75 randomized patients, clinical remission was maintained for 24 weeks in 27 (71%) of 38 patients in arm 1 (2 mg/kg every 4 weeks) and 31 (84%) of 37 patients in arm 2 (4 mg/kg every 8 weeks) (P ≤ 0.0001 for arm 1 versus arm 2 among those meeting the 40% threshold). Overall, 25 (33%) of 75 patients discontinued canakinumab, and clinical remission was maintained for at least 24 weeks in all 25 of these patients. No new safety signals were identified. CONCLUSION: Reduction of canakinumab exposure may be feasible in patients who have achieved clinical remission of systemic JIA, but consistent interleukin-1 inhibition appears necessary to maintain this response. ispartof: ARTHRITIS & RHEUMATOLOGY vol:73 issue:2 pages:336-346 ispartof: location:United States status: published

Published
2020

12. Population-based study of multisystem inflammatory syndrome associated with COVID-19 found that 36% of children had persistent symptoms

Author

Elisabet Berthold, Reza Rasti, Maria Altman, Charlotta Nordenhäll, Petter Brodin, Michele Compagno, Fatine Khammari Nystrom, Jenny Lingman Framme, Selma Idring Nordström, Erik Sundberg, Anders Fasth, Eva Säve-Söderbergh, Antti Kukka, Karin Palmblad, André Rudolph, Petra Król, Fredrik Bäckström, Robin Kahn, Karin Rydenman, Lillemor Berntson, Stefan Berg, Maria Mossberg, Bengt Månsson, Josefin Hättting, and AnnaCarin Horne

Subjects
medicine.medical_specialty, 2019-20 coronavirus outbreak, Coronavirus disease 2019 (COVID-19), Abnormal echocardiogram, Adolescent, Critical Care, persistent symptoms, outcomes, Pediatrics, World health, Internal medicine, Intensive care, medicine, Humans, Medical history, Child, abnormal echocardiograms, intensive care, Aged, business.industry, SARS-CoV-2, Pediatrik, COVID-19, General Medicine, Systemic Inflammatory Response Syndrome, Population based study, Echocardiography, Pediatrics, Perinatology and Child Health, fatigue, Abnormal results, business
Abstract

Aim: Our aim was to describe the outcomes of multisystem inflammatory syndrome in children (MIS-C) associated with COVID-19. Methods: This national, population-based, longitudinal, multicentre study used Swedish data that were prospectively collected between 1 December 2020 and 31 May 2021. All patients met the World Health Organization criteria for MIS-C. The outcomes 2 and 8 weeks after diagnosis are presented, and follow-up protocols are suggested. Results: We identified 152 cases, and 133 (87%) participated. When followed up 2 weeks after MIS-C was diagnosed, 43% of the 119 patients had abnormal results, including complete blood cell counts, platelet counts, albumin levels, electrocardiograms and echocardiograms. After 8 weeks, 36% of 89 had an abnormal patient history, but clinical findings were uncommon. Echocardiogram results were abnormal in 5% of 67, and the most common complaint was fatigue. Older children and those who received intensive care were more likely to report symptoms and have abnormal cardiac results. Conclusion: More than a third (36%) of the patients had persistent symptoms 8 weeks after MIS-C, and 5% had abnormal echocardiograms. Older age and higher levels of initial care appeared to be risk factors. Structured follow-up visits are important after MIS-C.

Published
2021

13. Tapering Canakinumab Monotherapy in Patients with Systemic Juvenile Idiopathic Arthritis in Clinical Remission: Results from an Open‐label, Randomized Phase IIIb/IV Study

Author

Pierre, Quartier, Ekaterina, Alexeeva, Constantin, Tamàs, Vyacheslav, Chasnyk, Nico, Wulffraat, Karin, Palmblad, Carine, Wouters, Hermine, Brunner, Katherine, Marzan, Rayfel, Schneider, Gerd, Horneff, Martini, Alberto, Jordi, Anton, Xiaoling, Wei, Alan, Slade, Ruperto, Nicolino, Ken, Abrams, Wolfgang, Emminger, Andrea, Ulbrich, Sugarka, Fodor, Lien, Desomer, Bernard, Lauwerys, Bénédicte, Brichard, Cécile, Boulanger, Gabriel, Levy, Laurence, Goffin, Phu Quoc Le, Marcia, Bandeira, Christina Feitosa Pelajo, Sheila Knupp Feitosa, Christianne, Costa, Marta Cristine Felix Rodrigues, Clovis Artur Almeida da Silva, Lucia Maria Mattei de, Katia, Kozu, Ronald, Laxer, Kristin, Houghton, Lori, Tucker, Kimberly, Morishita, Agnes, Mogenet, Richard, Mouy, Brigitte Bader Meunier, Candice, Meyzer, Michaela, Semeraro, Ouafa, Ben‐brahim, Isabelle, Kone‐paut, Caroline, Galeotti, Linda, Rossi, Perrine, Dusser, Bilade, Cherquaoui, Alexandre, Belot, Agnes, Duquesne, Freychet, Caroline, Laurent, Audrey, Marine, Desjonqueres, Ivan, Foeldvari, Antonia, Kienast, Barbara, Willig, Deborah, Barthel, Joachim, Peitz, Stefanie, Wintrich, Tilman Felix Geikowski, Anna Carina Schulz, Markus, Hufnagel, Marc, Hirdes, Rouven, Kubicki, Janbernd, Kirschner, Ales, Janda, Andre, Jacob, Cornelia, Emerich, Anna, Raab, Gonza, Ngoumou, Kirsten, Minden, Mareike, Lieber, Sae‐Lim von Stuckrad, Jasmin Kuemmerle Deschner, Sandra, Hansmann, Tom, Schleich, Ines Maria Magunia, Joachim, Riethmuller, Nicole, Anders, Hartwig, Lehmann, Jan de Laffolie, Thomas, Lutz, Juergen, Grulich‐henn, Johannes, Pfeil, Astrid, Helling‐bakki, Ralf, Trauzeddel, Daniel, Haselbusch, Henryk, Kolbeck, Elisabeth, Weissbarth‐riedel, Anja, Froehlich, Andrea, Ponyi, Diana, Garan, Ilonka, Orban, Krisztina, Sevcic, Yonatan, Butbul, Riva, Brik, Philip, Hashkes, Ori, Toker, Ruby, Haviv, Yosef, Uziel, Rubi, Haviv, Veronica, Moshe, Michal, Rothschild, Liora, Harel, Gil, Amarilyo, Rotem, Tal, Mohamad Hamad Said, Irit, Tirosh, Shiri, Spielman, Maya, Gerstein, Ravelli, Angelo, Schiappapietra, Benedetta, Varnier, GIULIA CAMILLA, Finetti, Martina, Marasini, Maurizio, Caorsi, Roberta, Rosina, Silvia, Federici, Silvia, Irene, Pontikaki, Pier Luigi Meroni, Valeri, Gerloni, Nicola, Ughi, Tania, Ubiali, Maria, Alessio, Roberto Della Casa, Sebastiaan Jozef Vastert, Joost Frans Swart, van Royen‐Kerhof, A., Ellen, Schatorje, Van Iperen‐Schutte, G., Lidia, Rutkowska‐sak, Izabela, Szczygielska, Malgorzata, Kwiatkowska, Maria, Marusak‐banacka, Piotr, Gietka, Kseniya, Isaeva, Rina, Denisova, Ludmila, Snegireva, Margarita, Dubko, Mikhail, Kostik, Natalia, Buchinskaia, Olga, Kalashnikova, Sergey, Avrusin, Vera, Masalova, Esmeralda Nunez Cuadros, Gisela, Diez, Rocio Galindo Zavala, Rosa Bou Torrent, Estibaliz, Iglesias, Joan, Calzada, Violeta, Bittermann, Alina Lucica Boteanu, Maria Luz Gamir, Inmaculada, Calvo, Berta, Lopez, Isabel, Gonzalez, Laura, Fernandez, Daniel Clemente Garulo, Juan Carlos Lopez Robledillo, Rosa, Merino, Rosa, Alcobendas, Agustin, Remesal, Sara, Murias, Magnusson, Bo, Ozgur, Kasapcopur, Kenan, Barut, Amra, Adrovic, Sezgin, Sahin, Muferet, Erguven, Refia Gozdenur Savci, Seza, Ozen, Selcan, Demir, Yelda, Bilginer, Zehra Serap Avci, Ezgi Deriz Batu, Andreas, Reiff, Anusha, Ramanatham, Diana, Brown, Bracha, Shaham, Shirley, Parks, Michal, Cidon, Gloria, Higgins, Charles, Spencer, Jenny, Rossette, Karla, Jones, Sharon Bout Tabaku, Shelli, Farley, Shoghik, Akoghlanian, Quartier, Pierre, Alexeeva, Ekaterina, Tamàs, Constantin, Chasnyk, Vyacheslav, Wulffraat, Nico, Palmblad, Karin, Wouters, Carine, Brunner, Hermine, Marzan, Katherine, Schneider, Rayfel, Horneff, Gerd, Martini, Alberto, Anton, Jordi, Wei, Xiaoling, Slade, Alan, Ruperto, Nicolino, Abrams, Ken, Emminger, Wolfgang, Ulbrich, Andrea, Fodor, Sugarka, Desomer, Lien, Lauwerys, Bernard, Brichard, Bénédicte, Boulanger, Cécile, Levy, Gabriel, Goffin, Laurence, Quoc Le, Phu, Bandeira, Marcia, Feitosa Pelajo, Christina, Knupp Feitosa, Sheila, Costa, Christianne, Cristine Felix Rodrigues, Marta, Artur Almeida da Silva, Clovi, Maria Mattei de, Lucia, Kozu, Katia, Laxer, Ronald, Houghton, Kristin, Tucker, Lori, Morishita, Kimberly, Mogenet, Agne, Mouy, Richard, Bader Meunier, Brigitte, Meyzer, Candice, Semeraro, Michaela, Ben‐brahim, Ouafa, Kone‐paut, Isabelle, Galeotti, Caroline, Rossi, Linda, Dusser, Perrine, Cherquaoui, Bilade, Belot, Alexandre, Duquesne, Agne, Caroline, Freychet, Audrey, Laurent, Desjonqueres, Marine, Foeldvari, Ivan, Kienast, Antonia, Willig, Barbara, Barthel, Deborah, Peitz, Joachim, Wintrich, Stefanie, Felix Geikowski, Tilman, Carina Schulz, Anna, Hufnagel, Marku, Hirdes, Marc, Kubicki, Rouven, Kirschner, Janbernd, Janda, Ale, Jacob, Andre, Emerich, Cornelia, Raab, Anna, Ngoumou, Gonza, Minden, Kirsten, Lieber, Mareike, von Stuckrad, Sae‐lim, Kuemmerle Deschner, Jasmin, Hansmann, Sandra, Schleich, Tom, Maria Magunia, Ine, Riethmuller, Joachim, Anders, Nicole, Lehmann, Hartwig, de Laffolie, Jan, Lutz, Thoma, Grulich‐henn, Juergen, Pfeil, Johanne, Helling‐bakki, Astrid, Trauzeddel, Ralf, Haselbusch, Daniel, Kolbeck, Henryk, Weissbarth‐riedel, Elisabeth, Froehlich, Anja, Ponyi, Andrea, Garan, Diana, Orban, Ilonka, Sevcic, Krisztina, Butbul, Yonatan, Brik, Riva, Hashkes, Philip, Toker, Ori, Haviv, Ruby, Uziel, Yosef, Haviv, Rubi, Moshe, Veronica, Rothschild, Michal, Harel, Liora, Amarilyo, Gil, Tal, Rotem, Hamad Said, Mohamad, Tirosh, Irit, Spielman, Shiri, Gerstein, Maya, Ravelli, Angelo, Schiappapietra, Benedetta, Camilla Varnier, Giulia, Finetti, Martina, Marasini, Maurizio, Caorsi, Roberta, Rosina, Silvia, Federici, Silvia, Pontikaki, Irene, Luigi Meroni, Pier, Gerloni, Valeri, Ughi, Nicola, Ubiali, Tania, Alessio, Maria, DELLA CASA, Roberto, Jozef Vastert, Sebastiaan, Frans Swart, Joost, van Royen‐Kerhof, A., Schatorje, Ellen, Van Iperen‐Schutte, G., Rutkowska‐sak, Lidia, Szczygielska, Izabela, Kwiatkowska, Malgorzata, Marusak‐banacka, Maria, Gietka, Piotr, Isaeva, Kseniya, Denisova, Rina, Snegireva, Ludmila, Dubko, Margarita, Kostik, Mikhail, Buchinskaia, Natalia, Kalashnikova, Olga, Avrusin, Sergey, Masalova, Vera, Nunez Cuadros, Esmeralda, Diez, Gisela, Galindo Zavala, Rocio, Bou Torrent, Rosa, Iglesias, Estibaliz, Calzada, Joan, Bittermann, Violeta, Lucica Boteanu, Alina, Luz Gamir, Maria, Calvo, Inmaculada, Lopez, Berta, Gonzalez, Isabel, Fernandez, Laura, Clemente Garulo, Daniel, Carlos Lopez Robledillo, Juan, Merino, Rosa, Alcobendas, Rosa, Remesal, Agustin, Murias, Sara, Magnusson, Bo, Kasapcopur, Ozgur, Barut, Kenan, Adrovic, Amra, Sahin, Sezgin, Erguven, Muferet, Gozdenur Savci, Refia, Ozen, Seza, Demir, Selcan, Bilginer, Yelda, Serap Avci, Zehra, Deriz Batu, Ezgi, Reiff, Andrea, Ramanatham, Anusha, Brown, Diana, Shaham, Bracha, Parks, Shirley, Cidon, Michal, Higgins, Gloria, Spencer, Charle, Rossette, Jenny, Jones, Karla, Bout Tabaku, Sharon, Farley, Shelli, and Akoghlanian, Shoghik

Published
2021

14. Tapering Canakinumab Monotherapy in Patients with Systemic Juvenile Idiopathic Arthritis in Clinical Remission: Results from an Open‐label, Randomized Phase IIIb/IV Study

Author

Quartier, P, Alexeeva, E, Tamàs, C, Chasnyk, V, Wulffraat, N, Palmblad, K, Wouters, C, Brunner, H, Marzan, K, Schneider, R, Horneff, G, Martini, A, Anton, J, Wei, X, Slade, A, Ruperto, N, Abrams, K, Emminger, W, Ulbrich, A, Fodor, S, Desomer, L, Lauwerys, B, Brichard, B, Boulanger, C, Levy, G, Goffin, L, Quoc Le, P, Bandeira, M, Feitosa Pelajo, C, Knupp Feitosa, S, Costa, C, Cristine Felix Rodrigues, M, Artur Almeida da Silva, C, Maria Mattei de, L, Kozu, K, Laxer, R, Houghton, K, Tucker, L, Morishita, K, Mogenet, A, Mouy, R, Bader Meunier, B, Meyzer, C, Semeraro, M, Ben‐brahim, O, Kone‐paut, I, Galeotti, C, Rossi, L, Dusser, P, Cherquaoui, B, Belot, A, Duquesne, A, Caroline, F, Audrey, L, Desjonqueres, M, Foeldvari, I, Kienast, A, Willig, B, Barthel, D, Peitz, J, Wintrich, S, Felix Geikowski, T, Carina Schulz, A, Hufnagel, M, Hirdes, M, Kubicki, R, Kirschner, J, Janda, A, Jacob, A, Emerich, C, Raab, A, Ngoumou, G, Minden, K, Lieber, M, von Stuckrad, S, Kuemmerle Deschner, J, Hansmann, S, Schleich, T, Maria Magunia, I, Riethmuller, J, Anders, N, Lehmann, H, de Laffolie, J, Lutz, T, Grulich‐henn, J, Pfeil, J, Helling‐bakki, A, Trauzeddel, R, Haselbusch, D, Kolbeck, H, Weissbarth‐riedel, E, Froehlich, A, Ponyi, A, Garan, D, Orban, I, Sevcic, K, Butbul, Y, Brik, R, Hashkes, P, Toker, O, Haviv, R, Uziel, Y, Moshe, V, Rothschild, M, Harel, L, Amarilyo, G, Tal, R, Hamad Said, M, Tirosh, I, Spielman, S, Gerstein, M, Ravelli, A, Schiappapietra, B, Camilla Varnier, G, Finetti, M, Marasini, M, Caorsi, R, Rosina, S, Federici, S, Pontikaki, I, Luigi Meroni, P, Gerloni, V, Ughi, N, Ubiali, T, Alessio, M, Della Casa, R, Jozef Vastert, S, Frans Swart, J, van Royen‐Kerhof, A, Schatorje, E, Van Iperen‐Schutte, G, Rutkowska‐sak, L, Szczygielska, I, Kwiatkowska, M, Marusak‐banacka, M, Gietka, P, Isaeva, K, Denisova, R, Snegireva, L, Dubko, M, Kostik, M, Buchinskaia, N, Kalashnikova, O, Avrusin, S, Masalova, V, Nunez Cuadros, E, Diez, G, Galindo Zavala, R, Bou Torrent, R, Iglesias, E, Calzada, J, Bittermann, V, Lucica Boteanu, A, Luz Gamir, M, Calvo, I, Lopez, B, Gonzalez, I, Fernandez, L, Clemente Garulo, D, Carlos Lopez Robledillo, J, Merino, R, Alcobendas, R, Remesal, A, Murias, S, Magnusson, B, Kasapcopur, O, Barut, K, Adrovic, A, Sahin, S, Erguven, M, Gozdenur Savci, R, Ozen, S, Demir, S, Bilginer, Y, Serap Avci, Z, Deriz Batu, E, Reiff, A, Ramanatham, A, Brown, D, Shaham, B, Parks, S, Cidon, M, Higgins, G, Spencer, C, Rossette, J, Jones, K, Bout Tabaku, S, Farley, S, Akoghlanian, S, Pierre Quartier, Ekaterina Alexeeva, Constantin Tamàs, Vyacheslav Chasnyk, Nico Wulffraat, Karin Palmblad, Carine Wouters, Hermine Brunner, Katherine Marzan, Rayfel Schneider, Gerd Horneff, Alberto Martini, Jordi Anton, Xiaoling Wei, Alan Slade, Nicolino Ruperto, Ken Abrams, Wolfgang Emminger, Andrea Ulbrich, Sugarka Fodor, Lien Desomer, Bernard Lauwerys, Bénédicte Brichard, Cécile Boulanger, Gabriel Levy, Laurence Goffin, Phu Quoc Le, Marcia Bandeira, Christina Feitosa Pelajo, Sheila Knupp Feitosa, Christianne Costa, Marta Cristine Felix Rodrigues, Clovis Artur Almeida da Silva, Lucia Maria Mattei de, Katia Kozu, Ronald Laxer, Kristin Houghton, Lori Tucker, Kimberly Morishita, Agnes Mogenet, Richard Mouy, Brigitte Bader Meunier, Candice Meyzer, Michaela Semeraro, Ouafa Ben‐Brahim, Isabelle Kone‐Paut, Caroline Galeotti, Linda Rossi, Perrine Dusser, Bilade Cherquaoui, Alexandre Belot, Agnes Duquesne, Freychet Caroline, Laurent Audrey, Marine Desjonqueres, Ivan Foeldvari, Antonia Kienast, Barbara Willig, Deborah Barthel, Joachim Peitz, Stefanie Wintrich, Tilman Felix Geikowski, Anna Carina Schulz, Markus Hufnagel, Marc Hirdes, Rouven Kubicki, Janbernd Kirschner, Ales Janda, Andre Jacob, Cornelia Emerich, Anna Raab, Gonza Ngoumou, Kirsten Minden, Mareike Lieber, Sae‐Lim von Stuckrad, Jasmin Kuemmerle Deschner, Sandra Hansmann, Tom Schleich, Ines Maria Magunia, Joachim Riethmuller, Nicole Anders, Hartwig Lehmann, Jan de Laffolie, Thomas Lutz, Juergen Grulich‐Henn, Johannes Pfeil, Astrid Helling‐Bakki, Ralf Trauzeddel, Daniel Haselbusch, Henryk Kolbeck, Elisabeth Weissbarth‐Riedel, Anja Froehlich, Andrea Ponyi, Diana Garan, Ilonka Orban, Krisztina Sevcic, Yonatan Butbul, Riva Brik, Philip Hashkes, Ori Toker, Ruby Haviv, Yosef Uziel, Rubi Haviv, Veronica Moshe, Michal Rothschild, Liora Harel, Gil Amarilyo, Rotem Tal, Mohamad Hamad Said, Irit Tirosh, Shiri Spielman, Maya Gerstein, Angelo Ravelli, Benedetta Schiappapietra, Giulia Camilla Varnier, Martina Finetti, Maurizio Marasini, Roberta Caorsi, Silvia Rosina, Silvia Federici, Irene Pontikaki, Pier Luigi Meroni, Valeri Gerloni, Nicola Ughi, Tania Ubiali, Maria Alessio, Roberto Della Casa, Sebastiaan Jozef Vastert, Joost Frans Swart, A. van Royen‐Kerhof, Ellen Schatorje, G. Van Iperen‐Schutte, Lidia Rutkowska‐Sak, Izabela Szczygielska, Malgorzata Kwiatkowska, Maria Marusak‐Banacka, Piotr Gietka, Kseniya Isaeva, Rina Denisova, Ludmila Snegireva, Margarita Dubko, Mikhail Kostik, Natalia Buchinskaia, Olga Kalashnikova, Sergey Avrusin, Vera Masalova, Esmeralda Nunez Cuadros, Gisela Diez, Rocio Galindo Zavala, Rosa Bou Torrent, Estibaliz Iglesias, Joan Calzada, Violeta Bittermann, Alina Lucica Boteanu, Maria Luz Gamir, Inmaculada Calvo, Berta Lopez, Isabel Gonzalez, Laura Fernandez, Daniel Clemente Garulo, Juan Carlos Lopez Robledillo, Rosa Merino, Rosa Alcobendas, Agustin Remesal, Sara Murias, Bo Magnusson, Ozgur Kasapcopur, Kenan Barut, Amra Adrovic, Sezgin Sahin, Muferet Erguven, Refia Gozdenur Savci, Seza Ozen, Selcan Demir, Yelda Bilginer, Zehra Serap Avci, Ezgi Deriz Batu, Andreas Reiff, Anusha Ramanatham, Diana Brown, Bracha Shaham, Shirley Parks, Michal Cidon, Gloria Higgins, Charles Spencer, Jenny Rossette, Karla Jones, Sharon Bout Tabaku, Shelli Farley, Shoghik Akoghlanian, Quartier, P, Alexeeva, E, Tamàs, C, Chasnyk, V, Wulffraat, N, Palmblad, K, Wouters, C, Brunner, H, Marzan, K, Schneider, R, Horneff, G, Martini, A, Anton, J, Wei, X, Slade, A, Ruperto, N, Abrams, K, Emminger, W, Ulbrich, A, Fodor, S, Desomer, L, Lauwerys, B, Brichard, B, Boulanger, C, Levy, G, Goffin, L, Quoc Le, P, Bandeira, M, Feitosa Pelajo, C, Knupp Feitosa, S, Costa, C, Cristine Felix Rodrigues, M, Artur Almeida da Silva, C, Maria Mattei de, L, Kozu, K, Laxer, R, Houghton, K, Tucker, L, Morishita, K, Mogenet, A, Mouy, R, Bader Meunier, B, Meyzer, C, Semeraro, M, Ben‐brahim, O, Kone‐paut, I, Galeotti, C, Rossi, L, Dusser, P, Cherquaoui, B, Belot, A, Duquesne, A, Caroline, F, Audrey, L, Desjonqueres, M, Foeldvari, I, Kienast, A, Willig, B, Barthel, D, Peitz, J, Wintrich, S, Felix Geikowski, T, Carina Schulz, A, Hufnagel, M, Hirdes, M, Kubicki, R, Kirschner, J, Janda, A, Jacob, A, Emerich, C, Raab, A, Ngoumou, G, Minden, K, Lieber, M, von Stuckrad, S, Kuemmerle Deschner, J, Hansmann, S, Schleich, T, Maria Magunia, I, Riethmuller, J, Anders, N, Lehmann, H, de Laffolie, J, Lutz, T, Grulich‐henn, J, Pfeil, J, Helling‐bakki, A, Trauzeddel, R, Haselbusch, D, Kolbeck, H, Weissbarth‐riedel, E, Froehlich, A, Ponyi, A, Garan, D, Orban, I, Sevcic, K, Butbul, Y, Brik, R, Hashkes, P, Toker, O, Haviv, R, Uziel, Y, Moshe, V, Rothschild, M, Harel, L, Amarilyo, G, Tal, R, Hamad Said, M, Tirosh, I, Spielman, S, Gerstein, M, Ravelli, A, Schiappapietra, B, Camilla Varnier, G, Finetti, M, Marasini, M, Caorsi, R, Rosina, S, Federici, S, Pontikaki, I, Luigi Meroni, P, Gerloni, V, Ughi, N, Ubiali, T, Alessio, M, Della Casa, R, Jozef Vastert, S, Frans Swart, J, van Royen‐Kerhof, A, Schatorje, E, Van Iperen‐Schutte, G, Rutkowska‐sak, L, Szczygielska, I, Kwiatkowska, M, Marusak‐banacka, M, Gietka, P, Isaeva, K, Denisova, R, Snegireva, L, Dubko, M, Kostik, M, Buchinskaia, N, Kalashnikova, O, Avrusin, S, Masalova, V, Nunez Cuadros, E, Diez, G, Galindo Zavala, R, Bou Torrent, R, Iglesias, E, Calzada, J, Bittermann, V, Lucica Boteanu, A, Luz Gamir, M, Calvo, I, Lopez, B, Gonzalez, I, Fernandez, L, Clemente Garulo, D, Carlos Lopez Robledillo, J, Merino, R, Alcobendas, R, Remesal, A, Murias, S, Magnusson, B, Kasapcopur, O, Barut, K, Adrovic, A, Sahin, S, Erguven, M, Gozdenur Savci, R, Ozen, S, Demir, S, Bilginer, Y, Serap Avci, Z, Deriz Batu, E, Reiff, A, Ramanatham, A, Brown, D, Shaham, B, Parks, S, Cidon, M, Higgins, G, Spencer, C, Rossette, J, Jones, K, Bout Tabaku, S, Farley, S, Akoghlanian, S, Pierre Quartier, Ekaterina Alexeeva, Constantin Tamàs, Vyacheslav Chasnyk, Nico Wulffraat, Karin Palmblad, Carine Wouters, Hermine Brunner, Katherine Marzan, Rayfel Schneider, Gerd Horneff, Alberto Martini, Jordi Anton, Xiaoling Wei, Alan Slade, Nicolino Ruperto, Ken Abrams, Wolfgang Emminger, Andrea Ulbrich, Sugarka Fodor, Lien Desomer, Bernard Lauwerys, Bénédicte Brichard, Cécile Boulanger, Gabriel Levy, Laurence Goffin, Phu Quoc Le, Marcia Bandeira, Christina Feitosa Pelajo, Sheila Knupp Feitosa, Christianne Costa, Marta Cristine Felix Rodrigues, Clovis Artur Almeida da Silva, Lucia Maria Mattei de, Katia Kozu, Ronald Laxer, Kristin Houghton, Lori Tucker, Kimberly Morishita, Agnes Mogenet, Richard Mouy, Brigitte Bader Meunier, Candice Meyzer, Michaela Semeraro, Ouafa Ben‐Brahim, Isabelle Kone‐Paut, Caroline Galeotti, Linda Rossi, Perrine Dusser, Bilade Cherquaoui, Alexandre Belot, Agnes Duquesne, Freychet Caroline, Laurent Audrey, Marine Desjonqueres, Ivan Foeldvari, Antonia Kienast, Barbara Willig, Deborah Barthel, Joachim Peitz, Stefanie Wintrich, Tilman Felix Geikowski, Anna Carina Schulz, Markus Hufnagel, Marc Hirdes, Rouven Kubicki, Janbernd Kirschner, Ales Janda, Andre Jacob, Cornelia Emerich, Anna Raab, Gonza Ngoumou, Kirsten Minden, Mareike Lieber, Sae‐Lim von Stuckrad, Jasmin Kuemmerle Deschner, Sandra Hansmann, Tom Schleich, Ines Maria Magunia, Joachim Riethmuller, Nicole Anders, Hartwig Lehmann, Jan de Laffolie, Thomas Lutz, Juergen Grulich‐Henn, Johannes Pfeil, Astrid Helling‐Bakki, Ralf Trauzeddel, Daniel Haselbusch, Henryk Kolbeck, Elisabeth Weissbarth‐Riedel, Anja Froehlich, Andrea Ponyi, Diana Garan, Ilonka Orban, Krisztina Sevcic, Yonatan Butbul, Riva Brik, Philip Hashkes, Ori Toker, Ruby Haviv, Yosef Uziel, Rubi Haviv, Veronica Moshe, Michal Rothschild, Liora Harel, Gil Amarilyo, Rotem Tal, Mohamad Hamad Said, Irit Tirosh, Shiri Spielman, Maya Gerstein, Angelo Ravelli, Benedetta Schiappapietra, Giulia Camilla Varnier, Martina Finetti, Maurizio Marasini, Roberta Caorsi, Silvia Rosina, Silvia Federici, Irene Pontikaki, Pier Luigi Meroni, Valeri Gerloni, Nicola Ughi, Tania Ubiali, Maria Alessio, Roberto Della Casa, Sebastiaan Jozef Vastert, Joost Frans Swart, A. van Royen‐Kerhof, Ellen Schatorje, G. Van Iperen‐Schutte, Lidia Rutkowska‐Sak, Izabela Szczygielska, Malgorzata Kwiatkowska, Maria Marusak‐Banacka, Piotr Gietka, Kseniya Isaeva, Rina Denisova, Ludmila Snegireva, Margarita Dubko, Mikhail Kostik, Natalia Buchinskaia, Olga Kalashnikova, Sergey Avrusin, Vera Masalova, Esmeralda Nunez Cuadros, Gisela Diez, Rocio Galindo Zavala, Rosa Bou Torrent, Estibaliz Iglesias, Joan Calzada, Violeta Bittermann, Alina Lucica Boteanu, Maria Luz Gamir, Inmaculada Calvo, Berta Lopez, Isabel Gonzalez, Laura Fernandez, Daniel Clemente Garulo, Juan Carlos Lopez Robledillo, Rosa Merino, Rosa Alcobendas, Agustin Remesal, Sara Murias, Bo Magnusson, Ozgur Kasapcopur, Kenan Barut, Amra Adrovic, Sezgin Sahin, Muferet Erguven, Refia Gozdenur Savci, Seza Ozen, Selcan Demir, Yelda Bilginer, Zehra Serap Avci, Ezgi Deriz Batu, Andreas Reiff, Anusha Ramanatham, Diana Brown, Bracha Shaham, Shirley Parks, Michal Cidon, Gloria Higgins, Charles Spencer, Jenny Rossette, Karla Jones, Sharon Bout Tabaku, Shelli Farley, and Shoghik Akoghlanian

Abstract

Objective: To evaluate the efficacy and safety of 2 canakinumab monotherapy tapering regimens in order to maintain complete clinical remission in children with systemic juvenile idiopathic arthritis (JIA). Methods: The study was designed as a 2-part phase IIIb/IV open-label, randomized trial. In the first part, patients received 4 mg/kg of canakinumab subcutaneously every 4 weeks and discontinued glucocorticoids and/or methotrexate as appropriate. Patients in whom clinical remission was achieved (inactive disease for at least 24 weeks) with canakinumab monotherapy were entered into the second part of the trial, in which they were randomized 1:1 into 1 of 2 treatment arms. In arm 1, the dose of canakinumab was reduced from 4 mg/kg to 2 mg/kg and then to 1 mg/kg, followed by discontinuation. In arm 2, the 4 mg/kg dose interval was prolonged from every 4 weeks, to every 8 weeks, and then to every 12 weeks, followed by discontinuation. In both arms, canakinumab exposure could be reduced provided systemic JIA remained in clinical remission for 24 weeks with each step. The primary objective was to assess whether >40% of randomized patients in either arm maintained clinical remission of systemic JIA for 24 weeks in the first part of the study. Results: In part 1 of the study, 182 patients were enrolled, with 75 of those patients randomized before entering part 2 of the trial. Among the 75 randomized patients, clinical remission was maintained for 24 weeks in 27 (71%) of 38 patients in arm 1 (2 mg/kg every 4 weeks) and 31 (84%) of 37 patients in arm 2 (4 mg/kg every 8 weeks) (P ≤ 0.0001 for arm 1 versus arm 2 among those meeting the 40% threshold). Overall, 25 (33%) of 75 patients discontinued canakinumab, and clinical remission was maintained for at least 24 weeks in all 25 of these patients. No new safety signals were identified. Conclusion: Reduction of canakinumab exposure may be feasible in patients who have achieved clinical remission of systemic JIA, but consisten

Published
2021

15. Expression of concern to: High systematic levels of the cytokine-inducing HMGB1 isoform secreted in severe macrophage activation syndrome

Author

Daniel J. Antoine, AnnaCarin Horne, Hanna Schierbeck, Erik Sundberg, Karin Palmblad, Helena Erlandsson Harris, Ulf Andersson, and Jan-Inge Henter

Subjects
Gene isoform, Expression of Concern, medicine.medical_treatment, lcsh:RM1-950, Biology, medicine.disease, HMGB1, Molecular medicine, lcsh:Biochemistry, Cytokine, lcsh:Therapeutics. Pharmacology, Macrophage activation syndrome, Genetics, medicine, Cancer research, biology.protein, Molecular Medicine, lcsh:QD415-436, Molecular Biology, Genetics (clinical)
Published
2020

16. Empowering Young People Living With Juvenile Idiopathic Arthritis to Better Communicate With Families and Care Teams: Content Analysis of Semistructured Interviews (Preprint)

Author

Stuart W Grande, Meghan R Longacre, Karin Palmblad, Meera V Montan, Rikard P Berquist, Andreas Hager, and Greg Kotzbauer

Abstract

BACKGROUND Young people living with juvenile idiopathic arthritis (JIA) face a number of communication barriers for achieving optimal health as they transition from pediatric care into adult care. Despite growing interest in mobile or wireless technologies to support health (mHealth), it is uncertain how these engagement tools might support young people, their families, and care teams to optimize preference-based treatment strategies. OBJECTIVE This study aims to examine how an mHealth patient support system (mPSS) might foster partnership between young people living with JIA, their families, and care teams. METHODS Semistructured interviews with young people (5-15 years old), their families, and JIA care teams were conducted using researcher-developed interviews guides. Transcribed data were qualitatively analyzed using conventional content analysis. RESULTS We conducted semistructured interviews with 15 young people, their parents, and 4 care team members. Content analysis revealed the potential of an mPSS to support productive dialogue between families and care teams. We identified four main themes: (1) young people with JIA face communication challenges, (2) normalizing illness through shared experience may improve adherence, (3) partnership opens windows into illness experiences, and (4) readiness to engage appears critical for clinic implementation. CONCLUSIONS A human-centered mPSS design that offers JIA patients the ability to track personally relevant illness concerns and needs can enhance communication, generate consensus-based treatment decisions, and improve efficiency and personalization of care. Technology that supports continuous learning and promotes better understanding of disease management may reduce practice burden while increasing patient engagement and autonomy in fostering lasting treatment decisions and ultimately supporting personalized care and improving outcomes.

Published
2018
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17. An in vivo cross-linkable hyaluronan gel with inherent anti-inflammatory properties reduces OA cartilage destruction in female mice subjected to cruciate ligament transection

Author

Karin Palmblad, H. Erlandsson Harris, Cecilia Aulin, Lars Klareskog, and P. Lundbäck

Subjects
0301 basic medicine, Cartilage, Articular, Anterior cruciate ligament, Biomedical Engineering, Anti-Inflammatory Agents, Osteoarthritis, Proinflammatory cytokine, Lipid peroxidation, 03 medical and health sciences, chemistry.chemical_compound, Mice, 0302 clinical medicine, Rheumatology, In vivo, medicine, Animals, Orthopedics and Sports Medicine, Anterior Cruciate Ligament, Hyaluronic Acid, 030203 arthritis & rheumatology, TUNEL assay, Cartilage, Anterior Cruciate Ligament Injuries, medicine.disease, Molecular biology, Hindlimb, Mice, Inbred C57BL, 030104 developmental biology, medicine.anatomical_structure, chemistry, Apoptosis, Immunology, Female, Gels
Abstract

Summary Objective To explore the possibility of cartilage protection in osteoarthritis (OA) by intraarticular injection of a chemically modified hyaluronan (HA) gel and investigate whether the chemical modifications provide intrinsic anti-inflammatory activity. Method OA was induced in C57BL/6 mice by anterior cruciate ligament transection (ACLT) and HA gel or carbazate-modified component was injected intra-articularly. Assessment of cartilage rescue was performed by histology, immunohistochemistry and TUNEL analysis. Serum levels of proinflammatory cytokines were evaluated with cytometric bead array, measuring IL-1β, TNF, IFN-γ, KC/CXCL1 and MCP-1. Results Intraarticular injection of the HA gel showed significantly reduced cartilage destruction and decreased osteophyte formation. Besides the biological and biomechanical effects of HA, we investigated lipid peroxidation products as an alternative inflammatory and potential mechanism contributing to OA. To address this, injection of the carbazate-modified component alone was performed, which also demonstrated a cartilage-saving effect. Besides the cartilage amelioration effects, decreased apoptosis, 4-hydroxynonenal (4-HNE) and MHC class II staining was recorded. No changes in serum levels of proinflammatory cytokines were detected. Conclusion We have shown that the HA gel has an anti-destructive effect on articular cartilage (AC). Our results demonstrated that the carbazate-modified component could suppress apoptotic events, potentially by quenching of ROS/LPO products such as 4-HNE in OA joints. Modification of the HA molecule offers opportunities to introduce (covalent) coupling of additional molecules to the gel, with controlled retention and subsequent release in the joint.

Published
2016

19. Empowering Young People Living With Juvenile Idiopathic Arthritis to Better Communicate With Families and Care Teams: Content Analysis of Semistructured Interviews

Author

Andreas Hager, Meera V Montan, Stuart W. Grande, Rikard P Berquist, Greg Kotzbauer, Meghan R Longacre, and Karin Palmblad

Subjects
Male, Adolescent, interviews, 020205 medical informatics, media_common.quotation_subject, Health Informatics, Information technology, 02 engineering and technology, Personalization, Interviews as Topic, 03 medical and health sciences, 0302 clinical medicine, Nursing, 0202 electrical engineering, electronic engineering, information engineering, Humans, health communication, Family, 030212 general & internal medicine, Disease management (health), Patient participation, Child, mHealth, Health communication, media_common, Patient Care Team, Sweden, Original Paper, Communication, Professional-Patient Relations, T58.5-58.64, Arthritis, Juvenile, juvenile arthritis, Content analysis, Child, Preschool, General partnership, Female, Family Relations, patient participation, Public aspects of medicine, RA1-1270, Psychology, Autonomy
Abstract

BackgroundYoung people living with juvenile idiopathic arthritis (JIA) face a number of communication barriers for achieving optimal health as they transition from pediatric care into adult care. Despite growing interest in mobile or wireless technologies to support health (mHealth), it is uncertain how these engagement tools might support young people, their families, and care teams to optimize preference-based treatment strategies. ObjectiveThis study aims to examine how an mHealth patient support system (mPSS) might foster partnership between young people living with JIA, their families, and care teams. MethodsSemistructured interviews with young people (5-15 years old), their families, and JIA care teams were conducted using researcher-developed interviews guides. Transcribed data were qualitatively analyzed using conventional content analysis. ResultsWe conducted semistructured interviews with 15 young people, their parents, and 4 care team members. Content analysis revealed the potential of an mPSS to support productive dialogue between families and care teams. We identified four main themes: (1) young people with JIA face communication challenges, (2) normalizing illness through shared experience may improve adherence, (3) partnership opens windows into illness experiences, and (4) readiness to engage appears critical for clinic implementation. ConclusionsA human-centered mPSS design that offers JIA patients the ability to track personally relevant illness concerns and needs can enhance communication, generate consensus-based treatment decisions, and improve efficiency and personalization of care. Technology that supports continuous learning and promotes better understanding of disease management may reduce practice burden while increasing patient engagement and autonomy in fostering lasting treatment decisions and ultimately supporting personalized care and improving outcomes.

Published
2019
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21. Cartilage-saving effects of local HMGB1-neutralizing therapy in experimental osteoarthritis

Author

Karin Palmblad, Helena Erlandsson Harris, L. Klareskog, and C. Aulin

Subjects
0301 basic medicine, Pathology, medicine.medical_specialty, biology, business.industry, Cartilage, Biomedical Engineering, Experimental osteoarthritis, HMGB1, 03 medical and health sciences, 030104 developmental biology, medicine.anatomical_structure, Rheumatology, medicine, biology.protein, Orthopedics and Sports Medicine, business
Published
2017
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