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1. Defining outcome measures in juvenile idiopathic arthritis associated uveitis by a systematic review analysis: do we need a consensus?

Author

Greta Mastrangelo, Ivan Foeldvari, Jordi Anton, and Gabriele Simonini

Subjects
Pediatrics, medicine.medical_specialty, lcsh:Diseases of the musculoskeletal system, Consensus, Visual acuity, genetic structures, Visual Acuity, Arthritis, Review, Disease, Amblyopia, Severity of Illness Index, Uveitis, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Quality of life, Adrenal Cortex Hormones, Internal medicine, Outcome Assessment, Health Care, Severity of illness, medicine, Humans, Immunology and Allergy, 030212 general & internal medicine, Outcome, 030203 arthritis & rheumatology, business.industry, lcsh:RJ1-570, lcsh:Pediatrics, Juvenile idiopathic arthritis, medicine.disease, Arthritis, Juvenile, Pediatrics, Perinatology and Child Health, Quality of Life, lcsh:RC925-935, medicine.symptom, business, Developed country, Immunosuppressive Agents
Abstract

Background Juvenile Idiopathic Arthritis associated Uveitis (JIA-U) represents its most frequent extra-articular manifestation and the main cause of childhood uveitis in in developed countries. The broad variety of outcome measures utilized makes the comparison of the disease course, risk for complications, impairment in visual function, and responses to treatment quite difficult. Our aim was to summarize evidence regarding the current availability of outcome measures in JIA-U. Methods A systematic review between January 2000 and December 2018 was performed to identify studies investigating outcome measures used in JIA-U. Results The initial search identified 8254 articles of which 89 were potentially eligible. After the full text revision, a total of 27 studies, including 2 RCTs, were included. Among these studies 12 outcome measures for JIA-U use have been identified (grade of cells in the AC, grade of flare in the AC, VA, amblyopia, structural complications, use and sparing of oral corticosteroids and immunosuppressive drugs, surgery requirement, biomarkers, bilateral disease, JIA persistence, quality of life assessments, uveitis subtype). As regards primary outcome measures, 44% among studies included one or more variables related to disease activity (i.e. grade of flare, grade of cells); 56% included visual function performance (i.e. visual acuity); 68% (17/25) included one or more variables of disease-associated tissue damage or complications (i.e. cataract, amblyopia); 24% included disease features (i.e. bilateral disease; uveitis subtype); 44% included laboratory features (i.e. biomarkers); 8% included JIA features (i.e. persistence of disease); 12% included quality of life (i.e. EYE-Q); 44% included management (i.e. use and sparing of oral corticosteroids and other immunosuppressive drugs; surgery requirement). Conclusions Our systematic review surveys the heterogeneity around outcome measures related to JIA-U in children, even in RCTs. It does not provide the solution to overcome the heterogeneity in uveitis studies, but it does provide an estimate of the scale of the problems and provides data to inform this important debate; highlighting the requirement to obtain a new consensus regarding a common approach to identify suitable and efficient outcome measures in JIA-U.

Published
2019
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2. Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part two

Author

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

Subjects
030203 arthritis & rheumatology, 03 medical and health sciences, medicine.medical_specialty, 0302 clinical medicine, Rheumatology, business.industry, 030220 oncology & carcinogenesis, Family medicine, Pediatrics, Perinatology and Child Health, medicine, Immunology and Allergy, business, Paediatric rheumatology
Published
2017
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3. SHARE – workpackage 5: development of best practices of diagnosis and treatment for paediatric rheumatic diseases throughout Europe

Author

Annet van Royen, Sebastiaan J. Vastert, Noortje Groot, Felicitas Belluti Enders, Tadej Avcin, Francesco Zulian, Pavla Dolezalova, Helene Foster, Alberto Martini, Sylvia Kamphuis, Michael W. Beresford, Angelo Ravelli, Roberta Culpo, Brian M. Feldman, Ivan Foeldvari, Nico M Wulffraat, and Liza J McCann

Subjects
medicine.medical_specialty, Pediatrics, business.industry, Treatment regimen, Best practice, Alternative medicine, Rheumatology, Internal medicine, Family medicine, Pediatrics, Perinatology and Child Health, Agency (sociology), Oral Presentation, Immunology and Allergy, Medicine, Pediatrics, Perinatology, and Child Health, Pediatric rheumatology, business
Abstract

Paediatric rheumatic diseases (PRD) form a group of (rare) diseases that can lead to significant morbidity. Evidence-based guidelines are sparse and treatment regimens differ throughout Europe. In 2012, a European initiative called SHARE (Single Hub and Access point for pediatric Rheumatology in Europe) was granted by the European Agency for Health and Consumers (project number 2011 1202) to optimize and disseminate diagnostic and management regimens in Europe for children with PRD.

Published
2014
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4. Uveitis outcome and complication

Author

Ivan Foeldvari

Subjects
musculoskeletal diseases, medicine.medical_specialty, Visual acuity, genetic structures, Arthritis, Disease, Rheumatology, Ophthalmology, Internal medicine, Immunology and Allergy, Medicine, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business.industry, Invited Speaker Presentation, Enthesitis, medicine.disease, Dermatology, eye diseases, Pediatrics, Perinatology and Child Health, Anterior uveitis, medicine.symptom, business, Complication, Uveitis
Abstract

Anterior uveitis is a well-known threatening comorbid condition of JIA and affects around 10 to 20 % of the patients depending on JIA subtype. A large proportion of children with JIA develop uveitis in the first year of disease and 73 to 90% do so after four years of the arthritis onset. Uveitis can progress into the adulthood and usually occur as “white uveitis” which is not associated with symptoms like redness and pain as opposed to JIA related to the enthesitis subtype that is symptomatic. Factors associated to lower uveitis remission rate are: JIA diagnosis, findings of 1+ or more vitreous cells at presentation and initial visual acuity of 20/200 or worse. The Standardization of Uveitis Nomenclature (SUN) Group took the first step to define outcome measures for uveitis, but it was established just for adults. The Multinational Interdisciplinary Working Group for Uveitis in Childhood (MIWGUC) proposed outcome measures for JIA associated uveitis incorporating the Standardization of Uveitis Nomenclature (SUN) criteria in 2011.

Published
2014
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5. SHARE – workpackage 5: evidence based recommendations for diagnosis and treatment of juvenile localized scleroderma and juvenile systemic sclerosis

Author

Roberta Culpo, Pavla Dolezalova, Jordi Anton, Claudia Saad-Magalhaes, Clarissa Pilkington, Jeffrey Chaitow, Sebastian J Vastert, Ivan Foeldvari, Annett van Royen, O Kasapcopur, Eileen Baildam, Tamas Constantin, Tadej Avcin, Ricardo Russo, Christina Boros, Sheila Knupp Feitosa de Oliveira, Angelo Ravelli, Nico M Wulffraat, Francesco Zulian, and N Toplak

Subjects
medicine.medical_specialty, Pediatrics, Evidence-based practice, Treatment regimen, business.industry, Alternative medicine, Rheumatology, Internal medicine, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Juvenile Localized Scleroderma, Immunology and Allergy, Juvenile, Pediatrics, Perinatology, and Child Health, Young adult, Pediatric rheumatology, business
Abstract

Juvenile Localized Scleroderma (JLS) and Juvenile Systemic Sclerosis (JSSc) form a group of rare pediatric diseases that can lead to significant morbidity. Evidence-based guidelines are sparse and management is mostly based on physician’s experience. Consequently, treatment regimens differ throughout Europe. In 2012, a European initiative called SHARE (Single Hub and Access point for pediatric Rheumatology in Europe) was launched to optimize and disseminate diagnostic and management regimens in Europe for children and young adults with rheumatic diseases.

Published
2014
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6. Methotrexate in oligoarticular persistent juvenile idiopathic arthritis

Author

A. Thon, Heinrike Schmeling, Gerd Ganser, Gerd Horneff, and Ivan Foeldvari

Subjects
musculoskeletal diseases, Poor prognosis, medicine.medical_specialty, genetic structures, business.industry, Arthritis, medicine.disease, eye diseases, Rheumatology, Disease activity, immune system diseases, Internal medicine, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Physical therapy, Immunology and Allergy, Juvenile, Methotrexate, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business, Glucocorticoid, medicine.drug
Abstract

Methotrexate (MTX) is recommended for children with juvenile idiopathic arthritis (JIA) with persistent oligoarticular course with high disease activity and features of poor prognosis or if they failed intraarticular glucocorticoid steroid injections following the 2011 ACR recommendations for the treatment of JIA. To date efficacy and safety of MTX for this JIA category have not been well studied.

Published
2014
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7. Results of a multinational survey regarding the diagnosis and treatment of temporomandibular joint involvement in juvenile idiopathic arthritis

Author

Nikolay Tzaribachev, Randy Q. Cron, and Ivan Foeldvari

Subjects
Male, musculoskeletal diseases, medicine.medical_specialty, Internationality, Short Report, Anti-Inflammatory Agents, MEDLINE, Arthritis, stomatognathic system, Rheumatology, Internal medicine, medicine, Humans, Mass Screening, Immunology and Allergy, Juvenile, Pediatrics, Perinatology, and Child Health, Disease management (health), Child, skin and connective tissue diseases, Glucocorticoids, Mass screening, Temporomandibular Joint, Tumor Necrosis Factor-alpha, business.industry, Data Collection, Anti-Inflammatory Agents, Non-Steroidal, Disease Management, Temporomandibular Joint Disorders, medicine.disease, Magnetic Resonance Imaging, Arthritis, Juvenile, Temporomandibular joint, Radiography, stomatognathic diseases, medicine.anatomical_structure, Antirheumatic Agents, Pediatrics, Perinatology and Child Health, Physical therapy, Female, Complication, business
Abstract

Background Temporomandibular joint (TMJ) involvement occurs in up to 80% of patients with juvenile idiopathic arthritis (JIA). Currently there are no standardized procedures regarding diagnosis and treatment of this common complication of JIA. The aim of the study was to assess the current clinical practices in many countries regarding diagnosis and treatment of TMJ involvement in JIA. Pediatric rheumatologists were asked to fill out a survey with 8 items regarding diagnosis and treatment of TMJ involvement. The survey was distributed over the worldwide pediatric rheumatology electronic list-serve. Data was collected in an Excel spread sheet and analyzed using Excel software. Findings Eighty-seven centers responded to the survey between December 2009 and April 2010. All responding centers were actively screening for TMJ involvement. All centers were screening by physical exam, 85 (97%) by history, and 2 (3%) by imaging. Seventy-seven (88%) centers were screening at the first visit and 76 (87%) at each follow-up visit. If imaging was requested, 77% of the centers reported that they asked for MRI, 10% for ultrasound, 9% for CT and 33% for X-ray. The first line treatment of TMJ arthritis was a non-biologic DMARD in 36%, an NSAID in 33%, an intraarticular corticosteroid injection in 26%, and an anti-TNF agent in 5%. Overall, 57 (65%) of the centers were using intraarticular corticosteroid injections as treatment. Conclusions TMJ arthritis is common among children with JIA. This survey shows that a wide array of diagnostic and therapeutic approaches is being employed for TMJ disease in 87 international centers. Due to this lack of agreement in how to diagnose and treat this JIA complication, we believe that an expert opinion/consensus statement regarding TMJ arthritis in JIA will likely benefit patients worldwide.

Published
2014
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8. PReS-FINAL-2126: How to follow up children with Raynaud syndrome - recommendations based on the Hamburg consensus meeting 2012

Author

I Konert, Clare E Pain, P. Hoeger, Dana Nemkova, M. Moll, Tamás Constantin, Nataša Toplak, Ivan Foeldvari, Smith, Daniela Gerent Petry Piotto, Nuray Aktay Ayaz, and Maurizio Cutolo

Subjects
medicine.medical_specialty, Pediatrics, business.industry, Systemic lupus, medicine.disease, Systemic scleroderma, Connective tissue disease, Rheumatology, immune system diseases, Internal medicine, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Immunology and Allergy, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business, Raynauds Phenomenon
Abstract

Raynauds phenomenon (RP) can be the first symptom of a connective tissue disease in children, in particular juvenile systemic scleroderma (JSSC) or systemic lupus erythematosis (SLE). However, the prevalence of RP in healthy school children has been shown to be as high as 15%[1]. There are currently no guidelines or agreed management strategies amongst Paediatric Rheumatologists on how to differentiate primary from secondary RP or how often patients require evaluation.

Published
2013
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9. Phase III, multi-centre, randomised, double blind, Placebo-controlled study for treatment of juvenile ankylosing spondylitis (AS) with Adalimumab

Author

K Kuemmerle-Deschner, G. Horneff, S Fitter, Rolf-Michael Kuester, Hans-Iko Huppertz, Michael Borte, Kirsten Minden, Ralf Trauzeddel, A. Thon, Gerd Ganser, Ivan Foeldvari, and N Tzaribacev

Subjects
medicine.medical_specialty, lcsh:Diseases of the musculoskeletal system, business.industry, Juvenile ankylosing spondylitis, lcsh:RJ1-570, Placebo-controlled study, lcsh:Pediatrics, Rheumatology, Double blind, Internal medicine, Ophthalmology, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Adalimumab, Immunology and Allergy, Pediatrics, Perinatology, and Child Health, lcsh:RC925-935, Multi centre, business, medicine.drug
Published
2011
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10. German evidence and consensus based guidelines 2010 for the treatment of juvenile idiopathic arthritis (JIA)

Author

H. Sitter, G. Horneff, Matthew P. Frosch, Martin Arbogast, Wolfram Singendonk, Hartmut Michels, Nihal Guellac, N. Wagner, Gerd Ganser, Matthias Schneider, Arnold Illhardt, G Dueckers, Marianne Spamer, Arnd Heiligenhaus, Ina Kopp, G Dannecker, Ivan Foeldvari, Ruediger Krauspe, Barbara Markus, and Tim Niehues

Subjects
medicine.medical_specialty, business.industry, Delphi method, Arthritis, medicine.disease, Rheumatology, language.human_language, German, Clinical trial, Family medicine, Internal medicine, Poster Presentation, Pediatrics, Perinatology and Child Health, Nominal group technique, medicine, language, Immunology and Allergy, Juvenile, Pediatrics, Perinatology, and Child Health, business, Scientific society
Abstract

Methods We performed a systematic literature analysis in PubMed (deadline: 15 January 2010, terms “juvenile idiopathic (rheumatoid) arthritis”, “therapy”, limits: “humans”, “published in the last 3 years”, “all child 018 years”, “clinical trial”) and evaluated relevant studies for quality of methodology. A formal consensus process, i.e. Nominal group technique and Delphi method, was conducted at three moderated consensus conferences at Dusseldorf or Krefeld (Germany) on May 9, 2007, August 1, 2007 and January 15, 2010. Conferences were attended by 95% of the representatives who had been nominated by their scientific society or organizations.

Published
2011
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11. Update of the results of a multinational survey regarding diagnosis and treatment of the temporomandibulare joint involvement in juvenile idiopathic arthritis- reflection of the day to day practice

Author

Nikolay Tzaribachev, Randy Q. Cron, Ivan Foeldvari, and Angela Wierk

Subjects
musculoskeletal diseases, medicine.medical_specialty, Pediatrics, lcsh:Diseases of the musculoskeletal system, genetic structures, Arthritis, stomatognathic system, Rheumatology, Internal medicine, medicine, Immunology and Allergy, Juvenile, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business.industry, lcsh:RJ1-570, lcsh:Pediatrics, medicine.disease, Temporomandibular joint, stomatognathic diseases, medicine.anatomical_structure, Joint involvement, Poster Presentation, Pediatrics, Perinatology and Child Health, lcsh:RC925-935, Day to day, business
Abstract

Temporomandibular joint (TMJ) involvement occurs up to 80% of patients with juvenile idiopathic arthritis (JIA). Currently they are no standardized procedures regarding diagnosis and treatment of this common presentation of JIA.

Published
2011
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12. Abatacept treatment improves health-related quality of life, pain, and sleep quality in juvenile idiopathic arthritis patients

Author

Edward H. Giannini, Christian Huemer, J. Chavez, N Ruperto, Michael Hofer, A Kivitz, Pierre Quartier, Ivan Foeldvari, Tracy Li, A Covucci, Alan J. Block, A Martini, F Blanco, Daniel J. Lovell, and Leonard H. Sigal

Subjects
musculoskeletal diseases, medicine.medical_specialty, lcsh:Diseases of the musculoskeletal system, Arthritis, Placebo, Child health, Rheumatology, Internal medicine, medicine, Immunology and Allergy, Juvenile, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, Health related quality of life, Sleep quality, business.industry, Abatacept, lcsh:RJ1-570, lcsh:Pediatrics, medicine.disease, Poster Presentation, Pediatrics, Perinatology and Child Health, Physical therapy, lcsh:RC925-935, business, medicine.drug
Abstract

Methods 190 JIA patients were treated with abatacept for 4 months in an open-label lead-in period (Period A). ACR Pedi 30 responders (n = 123) were then randomized 1:1 to receive abatacept or placebo for up to 6 months in a double-blind withdrawal period (Period B). HRQOL was assessed by the Child Health Questionnaire (CHQ), sleep quality was measured by the Children's Sleep Habits Questionnaire (CSHQ), and pain by a 0–100 mm VAS. Mean change from baseline in each period was calculated and compared between the treatment groups (in Period B), and the change over time was examined.

Published
2008
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13. First results of the uveitis outcome study of the multinational interdisciplinary working group for uveitis in childhood (MIWGUC)

Author

Valeria Gerloni, Kaisu Kotaniemi, Martina Niewerth, Eilsabeth Miserochchi, Kerstin Gerhold, Kirsten Minden, Susan Nielsen, Gabriele Brumm, C Delibero, Sandra Schenck, Gabriele Simonini, Ivan Foeldvari, Arnd Heiligenhaus, Uwe Pleyer, and I Sandfeldt

Subjects
musculoskeletal diseases, medicine.medical_specialty, Pediatrics, genetic structures, business.industry, Outcome measures, Arthritis, medicine.disease, Outcome (game theory), Prospective evaluation, Rheumatology, immune system diseases, Internal medicine, Ophthalmology, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Immunology and Allergy, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business, Uveitis
Abstract

Juvenile idiopathic arthritis (JIA) associated uveitis is one of the most severe comorbidities of JIA and it occurs in about 10 to 20 % of JIA patients. There are currently no specific established outcome measures for this specific uveitis. The Standardization of Uveitis Nomenclature SUN* group made the first attempt to establish outcome measures for uveitis in adults (1). We adapted part of it and developed and proposed outcome measures over a consensus process specific for JIA associated uveitis (2). Here we present the first results of the prospective evaluation of this outcome measures.

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14. When is a autologous bone marrow transplantation indicated in the treatment of juvenile systemic sclerorsis? Results of a multinational survey of Pediatric Rheumatologist

Author

Dominique Farge, Angela Wierk, and Ivan Foeldvari

Subjects
medicine.medical_specialty, Pediatrics, lcsh:Diseases of the musculoskeletal system, Ejection fraction, Cyclophosphamide, business.industry, lcsh:RJ1-570, lcsh:Pediatrics, Rheumatology, Transplantation, Therapeutic approach, FEV1/FVC ratio, DLCO, Internal medicine, Poster Presentation, Pediatrics, Perinatology and Child Health, medicine, Immunology and Allergy, Rituximab, Pediatrics, Perinatology, and Child Health, lcsh:RC925-935, business, medicine.drug
Abstract

The five year survival of juvenile systemic sclerosis (jSSc) is around 95%. Patients, who died in the two retrospective cohorts, died mostly in the first 24 months of disease course. Autologous bone marrow transplantation (ABMT) seems to be a promising therapeutic approach for adult patients ( ASTIS (EU) and SCOT (USA)Trial) with severe disease course. Around 8 patients with jSSc are transplanted according the EBMT registry. Currently no consent based inclusion or exclusion criteria for ABMT in jSSc exists. Aim of the survey was to get a feeling from paediatric rheumatologists, when they would apply autologous bone marrow transplantation as a treatment option. Paediatric Rheumatologist - members of the PRES Juvenile Scleroderma Working Group and participants of the Paediatric Rheumatology E-mail Board were asked via Internet to fill out the survey 22 centres responded, all of them were academic centres. BMT would be considered for 14 of the 22 colleagues after nonresponse to cyclophosphamide, 10 of 22 after non response to two DMARDs and 12 of 22 after nonresponse to Rituximab. 19 of 22 would consider transplantation if the CHAQ score ≤ 2 , 20 of 22 if the CHQ is less than 40%. 21 of 22 would think about transplantation if the modified Rodnan skin score is more than 30 and 15 of 22 if the DLCO is less than 50%, 18 of 22 if the WHO functional class is 3, 14 of 22 if the FVC less than 60%, 15 of 22 if the pulmonary arterial pressure more than 40 mm/hg and 11 of 22 if left ventricular ejection fraction is less than 40%. This survey represents an impression, when pediatric rheumatologist would consider ABMT. It is a starting point for a possible evolving ABMT program for this orphan disease

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15. PReS-FINAL-2063: Proposed criteria for activity, damage and impact of juvenile idiopathic arthritis associated uveitis: consensus effort from the multinational interdisciplinary working group for uveitis in childhood (MIWGUC)

Author

Kaisu Kotaniemi, R Ramanan, Susan Nielsen, E Rabinovich, Clive Edelsten, J. Anton, Ivan Foeldvari, Arnd Heiligenhaus, Bahram Bodaghi, and T. Saurenmann

Subjects
musculoskeletal diseases, Pediatrics, medicine.medical_specialty, Blinding, genetic structures, Childhood arthritis, business.industry, food and beverages, Arthritis, medicine.disease, Rheumatology, immune system diseases, Internal medicine, Ophthalmology, Pediatrics, Perinatology and Child Health, Poster Presentation, medicine, Juvenile, Immunology and Allergy, Pediatrics, Perinatology, and Child Health, skin and connective tissue diseases, business, Uveitis
Abstract

Uveitis is the most common extraartricular manifesation of juvenile idiopathic arthritis (JIA) with an.incidence of approx. 10 - 15 per 100,000 children. Uveitis, a potentially blinding disorder, is present in approx 10-18% of JIA patients, with a wide spectrum of presentations. Despite advances in treatment of childhood arthritis there are no definitions for assessment of activity, damage and impact of JIA associated uveitis.

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16. Update on the juvenile systemic sclerosis inception cohort http://www.juvenile-scleroderma.com

Author

Katsikas M, Dana Nemcova, Kallnich T, M. Kostic, K. Minden, M. Moll, Rolando Cimaz, Ivan Foeldvari, Juergen Brunner, A. Wierk, M. T. Terreri, Flavio Sztajnbok, and T. Avcin

Subjects
Gerontology, Text mining, Rheumatology, business.industry, Poster Presentation, Pediatrics, Perinatology and Child Health, Medicine, Juvenile, Immunology and Allergy, Pediatrics, Perinatology, and Child Health, business, Bioinformatics, INCEPTION COHORT
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