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152. Lyme-Arthritis

Author

Horneff, Gerd, Wahn, Volker, Reinhardt, Dietrich, editor, Nicolai, Thomas, editor, and Zimmer, Klaus-Peter, editor

Published
2014
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155. Efficacy and safety of tocilizumab in patients with polyarticular-course juvenile idiopathic arthritis: results from a phase 3, randomised, double-blind withdrawal trial

Author

Brunner, Hermine I, Ruperto, Nicolino, Zuber, Zbigniew, Keane, Caroline, Harari, Olivier, Kenwright, Andrew, Lu, Peng, Cuttica, Ruben, Keltsev, Vladimir, Xavier, Ricardo M, Calvo, Inmaculada, Nikishina, Irina, Rubio-Pérez, Nadina, Alexeeva, Ekaterina, Chasnyk, Vyacheslav, Horneff, Gerd, Opoka-Winiarska, Violetta, Quartier, Pierre, Silva, Clovis A, Silverman, Earl, Spindler, Alberto, Baildam, Eileen, Gámir, M Luz, Martin, Alan, Rietschel, Christoph, Siri, Daniel, Smolewska, Elzbieta, Lovell, Daniel, Martini, Alberto, De Benedetti, Fabrizio, Espada, Graciela, Allen, Roger, Chaitow, Jeffrey, Joos, Rik, Wouters, Carine, Knupp, Sheila, Sztajnbok, Flavio, Cabral, David, Houghton, Kristin, Roth, Johannes, Schmeling, Heinrike, Job-Deslandre, Chantal, Jorgensen, Christian, Paut, Isabelle Kone, Minden, Kirsten, Weller-Heinemann, Frank, Gerloni, Valeria, Zulian, Francesco, Burgos-Vargas, Ruben, Salazar, Carolina Duarte, Solis-Vallejo, Eunice, Calvo, Armando, Chavez, José, Zavaler, Manuel Ferrandiz, Gruenpeter, Anna, Kobusinska, Katarzyna, Sarychev, Alexey, Zholobova, Elena, Ramanan, Athimalaipet, Woo, Patricia, Goodman, Steven, Gedalia, Abraham, Kimura, Yukiko, Onel, Karen, and Schikler, Kenneth

Published
2015
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158. Consumer perspective on healthcare services for juvenile idiopathic arthritis: results of a multicentre JIA inception cohort study

Author

Heinrich-Rohr, Michaela, primary, Moenkemoeller, Kirsten, additional, Niewerth, Martina, additional, Sengler, Claudia, additional, Liedmann, Ina, additional, Kallinich, Tilmann, additional, Horneff, Gerd, additional, Windschall, Daniel, additional, Haas, Johannes-Peter, additional, Dressler, Frank, additional, Foeldvari, Ivan, additional, Weller-Heinemann, Frank, additional, Hospach, Toni, additional, Kuemmerle-Deschner, Jasmin, additional, Foell, Dirk, additional, Klotsche, Jens, additional, and Minden, Kirsten, additional

Published
2021
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159. Initial presenting manifestations in 16,486 patients with inborn errors of immunity include infections and noninfectious manifestations

Author

Thalhammer, Julian, primary, Kindle, Gerhard, additional, Nieters, Alexandra, additional, Rusch, Stephan, additional, Seppänen, Mikko R.J., additional, Fischer, Alain, additional, Grimbacher, Bodo, additional, Edgar, David, additional, Buckland, Matthew, additional, Mahlaoui, Nizar, additional, Ehl, Stephan, additional, Boztug, Kaan, additional, Brunner, Juergen, additional, Demel, Ulrike F., additional, Förster-Waldl, Elisabeth, additional, Gasteiger, Lukas M., additional, Göschl, Lisa, additional, Kojić, Marina, additional, Schroll, Andrea, additional, Seidel, Markus G., additional, Wintergerst, Uwe, additional, Wisgrill, Lukas, additional, Sharapova, Svetlana O., additional, Goffard, Jean-Christophe, additional, Kerre, Tessa, additional, Meyts, Isabelle, additional, Roosens, Fine, additional, Smet, Julie, additional, Haerynck, Filomeen, additional, Eric, Zelimir Pavle, additional, Milenova, Veneta, additional, Gagro, Alenka, additional, Richter, Darko, additional, Chovancova, Zita, additional, Hlavackova, Eva, additional, Litzman, Jiri, additional, Milota, Tomas, additional, Sediva, Anna, additional, Elaziz, Dalia Abd, additional, Alkady, Radwa Salaheldin, additional, El Sayed El Hawary, Rabab, additional, Eldash, Alia S., additional, Galal, Nermeen, additional, Lotfy, Sohilla, additional, Meshaal, Safa S., additional, Reda, Shereen M., additional, Sobh, Ali, additional, Elmarsafy, Aisha, additional, Brosselin, Pauline, additional, Courteille, Virginie, additional, De Vergnes, Nathalie, additional, Kracker, Sven, additional, Pergent, Martine, additional, Randrianomenjanahary, Philippe, additional, Ahrenstorf, Gerrit, additional, Albert, Michael H., additional, Ankermann, Tobias, additional, Atschekzei, Faranaz, additional, Baumann, Ulrich, additional, Becker, Benjamin C., additional, Behrends, Uta, additional, Belohradsky, Bernd H., additional, Biegner, Anika-Kerstin, additional, Binder, Nadine, additional, Bode, Sebastian F.N., additional, Boesecke, Christoph, additional, Boetticher, Benedikt, additional, Borte, Michael, additional, Borte, Stephan, additional, Classen, Carl Friedrich, additional, Dirks, Johannes, additional, Dückers, Gregor, additional, El-Helou, Sabine, additional, Ernst, Diana, additional, Fasshauer, Maria, additional, Fecker, Gisela, additional, Felgentreff, Kerstin, additional, Foell, Dirk, additional, Ghosh, Sujal, additional, Girschick, Hermann J., additional, Goldacker, Sigune, additional, Graf, Norbert, additional, Graf, Dagmar, additional, Greil, Johann, additional, Hanitsch, Leif Gunnar, additional, Hauck, Fabian, additional, Heeg, Maximilian, additional, Heine, Sabine I., additional, Henes, Joerg C., additional, Hoenig, Manfred, additional, Holzer, Ursula, additional, Holzinger, Dirk, additional, Horneff, Gerd, additional, Hundsdoerfer, Patrick, additional, Jablonka, Alexandra, additional, Jakoby, Donate, additional, Joean, Oana, additional, Kaiser-Labusch, Petra, additional, Klemann, Christian, additional, Kobbe, Robin, additional, Körholz, Julia, additional, Kramm, Christof M., additional, Krüger, Renate, additional, Landwehr-Kenzel, Sybille, additional, Lehmberg, Kai, additional, Liese, Johannes G., additional, Lippert, Conrad Ferdinand, additional, Maccari, Maria Elena, additional, Masjosthusmann, Katja, additional, Meinhardt, Andrea, additional, Metzler, Markus, additional, Morbach, Henner, additional, Müller, Ingo, additional, Naumann-Bartsch, Nora, additional, Neubert, Jennifer, additional, Niehues, Tim, additional, Peter, Hans-Hartmut, additional, Rieber, Nikolaus, additional, Ritterbusch, Henrike, additional, Rockstroh, Jürgen Kurt, additional, Roesler, Joachim, additional, Schauer, Uwe, additional, Scheible, Raphael, additional, Schmalzing, Marc, additional, Schmidt, Reinhold Ernst, additional, Schneider, Dominik T., additional, Schreiber, Stefan, additional, Schuetz, Catharina, additional, Schulz, Ansgar, additional, Schulze-Koops, Hendrik, additional, Schulze-Sturm, Ulf, additional, Schuster, Volker, additional, Schwaneck, Eva C., additional, Schwarz, Klaus, additional, Schwarze-Zander, Carolynne, additional, Sirin, Mehtap, additional, Skapenko, Alla, additional, Sogkas, Georgios, additional, Sparber-Sauer, Monika, additional, Speckmann, Carsten, additional, Steinmann, Sandra, additional, Stiehler, Sophie, additional, Tenbrock, Klaus, additional, von Bernuth, Horst, additional, Warnatz, Klaus, additional, Wasmuth, Jan-Christian, additional, Weiss, Michael, additional, Witte, Torsten, additional, Wittke, Kirsten, additional, Wittkowski, Helmut, additional, Zeuner, Rainald A., additional, Farmaki, Evangelia, additional, Hatzistilianou, Maria N., additional, Kakkas, Ioannis, additional, Kanariou, Maria G., additional, Kapousouzi, Androniki, additional, Liatsis, Emmanouil, additional, Maggina, Paraskevi, additional, Papadopoulou-Alataki, Efimia, additional, Raptaki, Maria, additional, Speletas, Matthaios, additional, Tantou, Sofia, additional, Goda, Vera, additional, Kriván, Gergely, additional, Marodi, Laszlo, additional, Abolhassani, Hassan, additional, Aghamohammadi, Asghar, additional, Rezaei, Nima, additional, Feighery, Conleth, additional, Leahy, Timothy Ronan, additional, Ryan, Paul, additional, Batzir, Nurit Assia, additional, Garty, Ben Zion, additional, Tamary, Hannah, additional, Aiuti, Alessandro, additional, Amodio, Donato, additional, Azzari, Chiara, additional, Barzaghi, Federica, additional, Baselli, Lucia A., additional, Cancrini, Caterina, additional, Carrabba, Maria, additional, Cazzaniga, Marco, additional, Cesaro, Simone, additional, Chinello, Matteo, additional, Danieli, Maria Giovanna, additional, Dellepiane, Rosa Maria, additional, Fabio, Giovanna, additional, Gambineri, Eleonora, additional, Lodi, Lorenzo, additional, Lougaris, Vassilios, additional, Marasco, Carolina, additional, Martire, Baldassarre, additional, Marzollo, Antonio, additional, Milito, Cinzia, additional, Moschese, Viviana, additional, Pignata, Claudio, additional, Plebani, Alessandro, additional, Porta, Fulvio, additional, Quinti, Isabella, additional, Ricci, Silvia, additional, Soresina, Annarosa, additional, Tommasini, Alberto, additional, Vacca, Angelo, additional, Vanessa, Clementina, additional, Blažienė, Audra, additional, Sitkauskiene, Brigita, additional, Gowin, Ewelina, additional, Heropolitańska-Pliszka, Edyta, additional, Pietrucha, Barbara, additional, Szaflarska, Anna, additional, Więsik-Szewczyk, Ewa, additional, Wolska-Kuśnierz, Beata, additional, Esteves, Isabel, additional, Faria, Emilia, additional, Marques, Laura Hora, additional, Neves, João Farela, additional, Silva, Susana L., additional, Teixeira, Carla, additional, Pereira da Silva, Sara, additional, Capilna, Brindusa Ruxandra, additional, Guseva, Marina N., additional, Shcherbina, Anna, additional, Bobcakova, Anna, additional, Ciznar, Peter, additional, Gabzdilova, Juliana, additional, Jesenak, Milos, additional, Kapustova, Lenka, additional, Orosova, Jaroslava, additional, Petrovicova, Otilia, additional, Raffac, Stefan, additional, Kopač, Peter, additional, Allende, Luis M., additional, Antolí, Arnau, additional, Blanch, Gemma Rocamora, additional, Carbone, Javier, additional, Dieli-Crimi, Romina, additional, Garcia-Prat, Marina, additional, Gil-Herrera, Juana, additional, Gonzalez-Granado, Luis Ignacio, additional, Agulló, Pilar Llobet, additional, Olbrich, Peter, additional, Parra-Martínez, Alba, additional, Paz-Artal, Estela, additional, Pleguezuelo, Daniel E., additional, Rodríguez, Nerea Salmón, additional, Sánchez-Ramón, Silvia, additional, Santos-Pérez, Juan Luis, additional, Solanich, Xavier, additional, Soler-Palacin, Pere, additional, González-Amores, Miriam, additional, Ekwall, Olov, additional, Fasth, Anders, additional, Bitzenhofer-Grüber, Michaela, additional, Candotti, Fabio, additional, Dimitriou, Florentia, additional, Heininger, Ulrich, additional, Holbro, Andreas, additional, Jandus, Peter, additional, Kolios, Antonios G.A., additional, Marschall, Karin, additional, Schmid, Jana Pachlopnik, additional, Posfay-Barbe, Klara M., additional, Prader, Seraina, additional, Reichenbach, Janine, additional, Steiner, Urs C., additional, Trück, Johannes, additional, Bredius, Robbert G., additional, de Kruijf- Bazen, Suzanne, additional, de Vries, Esther, additional, Henriet, Stefanie S.V., additional, Kuijpers, Taco W., additional, Potjewijd, Judith, additional, Rutgers, Abraham, additional, Stol, Kim, additional, van Aerde, Koen J., additional, Van den Berg, J. Merlijn, additional, van de Ven, Annick A.J.M., additional, Montfrans, Jorisvan, additional, Aydemir, Sezin, additional, Baris, Safa, additional, Dogu, Figen, additional, Ikinciogullari, Aydan, additional, Karakoc-Aydiner, Elif, additional, Kilic, Sara S., additional, Kiykim, Ayca, additional, Kökçü Karadağ, Şefika İlknur, additional, Kutukculer, Necil, additional, Ocak, Suheyla, additional, UNAL, Ekrem, additional, Boyarchuk, Oksana, additional, Hilfanova, Anna, additional, Kostyuchenko, Larysa V., additional, Alachkar, Hana, additional, Arkwright, Peter D., additional, Baxendale, Helen E., additional, Bernatoniene, Jolanta, additional, Coulter, Tanya I., additional, Garcez, Tomaz, additional, Goddard, Sarah, additional, Gompels, Mark M., additional, Grigoriadou, Sofia, additional, Herriot, Richard, additional, Herwadkar, Archana, additional, Huissoon, Aarnoud, additional, Ibberson, Lisa, additional, Nademi, Zoreh, additional, Noorani, Sadia, additional, Parvin, Shahnaz, additional, Steele, Cathal Laurence, additional, Thomas, Moira, additional, Waruiru, Catherine, additional, Yong, Patrick F.K., additional, and Bourne, Helen, additional

Published
2021
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169. Efficacy and safety of open-label etanercept on extended oligoarticular juvenile idiopathic arthritis, enthesitis-related arthritis and psoriatic arthritis: part 1 (week 12) of the CLIPPER study

Author

Horneff, Gerd, Burgos-Vargas, Ruben, Constantin, Tamas, Foeldvari, Ivan, Vojinovic, Jelena, Chasnyk, Vyacheslav G, Dehoorne, Joke, Panaviene, Violeta, Susic, Gordana, Stanevica, Valda, Kobusinska, Katarzyna, Zuber, Zbigniew, Mouy, Richard, Rumba-Rozenfelde, Ingrida, Breda, Luciana, Dolezalova, Pavla, Job-Deslandre, Chantal, Wulffraat, Nico, Alvarez, Daniel, Zang, Chuanbo, Wajdula, Joseph, Woodworth, Deborah, Vlahos, Bonnie, Martini, Alberto, and Ruperto, Nicolino

Published
2014
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170. Additional file 1 of Re-treatment with etanercept is as effective as the initial firstline treatment in patients with juvenile idiopathic arthritis

Author

Klotsche, Jens, Klein, Ariane, Niewerth, Martina, Hoff, Paula, Windschall, Daniel, Foeldvari, Ivan, Johannes-Peter Haas, Horneff, Gerd, and Minden, Kirsten

Subjects
musculoskeletal diseases, skin and connective tissue diseases
Abstract

Additional file 1: Supplemental Table 1. Univariable correlates of etanercept withdrawal after achieving inactive disease.

Published
2021
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171. miR-23a contributes to T cellular redox metabolism in juvenile idiopathic oligoarthritis

Author

Rajendiran, Anandhi, primary, Klemm, Patricia, additional, Schippers, Anastasia, additional, Scheufen, Anja, additional, Schwarz, Tobias, additional, Peitz, Joachim, additional, Brandenburg, Lars-Ove, additional, Wagner, Norbert, additional, Consolaro, Alessandro, additional, Raggi, Federica, additional, Bosco, Maria Carla, additional, Luedde, Tom, additional, Foell, Dirk, additional, Denecke, Bernd, additional, Horneff, Gerd, additional, Ohl, Kim, additional, and Tenbrock, Klaus, additional

Published
2021
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172. MRP8/14 serum levels as diagnostic markers for systemic juvenile idiopathic arthritis in children with prolonged fever

Author

Park, Carolin, primary, Miranda-Garcia, María, additional, Berendes, Rainer, additional, Horneff, Gerd, additional, Kuemmerle-Deschner, Jasmin, additional, Ganser, Gerd, additional, Huppertz, Hans-Iko, additional, Minden, Kirsten, additional, Haas, Johannes-Peter, additional, Jansson, Annette F, additional, Borte, Michael, additional, Schuetz, Catharina, additional, Oommen, Prasad, additional, Frosch, Michael, additional, Schlueter, Bernhard, additional, Richter-Unruh, Annette, additional, Kessel, Christoph, additional, Hinze, Claas, additional, Wittkowski, Helmut, additional, Roth, Johannes, additional, Foell, Dirk, additional, and Holzinger, Dirk, additional

Published
2021
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175. Clinical manifestations and outcome of SARS-CoV-2 infections in children and adolescents with rheumatic musculoskeletal diseases: data from the National Paediatric Rheumatology Database in Germany

Author

Sengler, Claudia, primary, Eulert, Sascha, additional, Minden, Kirsten, additional, Niewerth, Martina, additional, Horneff, Gerd, additional, Kuemmerle-Deschner, Jasmin, additional, Siemer, Caroline, additional, Berendes, Rainer, additional, Girschick, Hermann, additional, Hühn, Regina, additional, Borte, Michael, additional, Hospach, Anton, additional, Emminger, Wolfgang, additional, Armann, Jakob, additional, Klein, Ariane, additional, and Kallinich, Tilmann, additional

Published
2021
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177. Adalimumub with or without Methotrexate in juvenile rheumatoid arthritis

Author

Lovell, Daniel J., Ruperto, Nicolino, Goodman, Steven, Reiff, Andreas, Jung, Lawrence, Jarosova, Katerina, Nemcova, Dana, Muoy, Richard, Sandborg, Christy, Bohnsack, John, Elewaut, Dirk, Foeldvari, Ivan, Gerloni, Valeria, Rovensky, Jozef, Minden, Kirsten, Vehe, Richard K., Weiner, L. Wagner, Horneff, Gerd, Huppertz, Hans-Iko, Olson, Nancy Y., Medich, John R., Carcereri-De-Prati, Roberto, Mcllraith, Melissa J., Giannini, Edward H., and Martini, Alberto

Subjects
Rheumatoid arthritis in children -- Drug therapy, Tumor necrosis factor -- Research
Abstract

A study was conducted to determine the efficacy and safety of adalimumab in children with polyarticular-course juvenile rheumatoid arthritis. Results indicated that adalimumab was quite effective and safe as a form of treatment in children with juvenile rheumatoid arthritis.

Published
2008

180. A distinct CD38(+)CD45RA(+) population of CD4(+), CD8(+), and double-negative T cells is controlled by FAS

Author

Maccari, Maria Elena, Fuchs, Sebastian, Kury, Patrick, Andrieux, Geoffroy, Volkl, Simon, Bengsch, Bertram, Lorenz, Myriam Ricarda, Heeg, Maximilian, Rohr, Jan, Jagle, Sabine, Castro, Carla N., Gross, Miriam, Warthorst, Ursula, Koenig, Christoph, Fuchs, Ilka, Speckmann, Carsten, Thalhammer, Julian, Kapp, Friedrich G., Seidel, Markus G., Duckers, Gregor, Schoenberger, Stefan, Schuetz, Catharina, Fuhrer, Marita, Kobbe, Robin, Holzinger, Dirk, Klemann, Christian, Smisek, Petr, Owens, Stephen, Horneff, Gerd, Kolb, Reinhard, Naumann-Bartsch, Nora, Miano, Maurizio, Staniek, Julian, Rizzi, Marta, Kalina, Tomas, Schneider, Pascal, Erxleben, Anika, Backofen, Rolf, Ekici, Arif, Niemeyer, Charlotte M., Warnatz, Klaus, Grimbacher, Bodo, Eibel, Hermann, Mackensen, Andreas, Frei, Andreas Philipp, Schwarz, Klaus, Boerries, Melanie, Ehl, Stephan, Rensing-Ehl, Anne, Maccari, Maria Elena, Fuchs, Sebastian, Kury, Patrick, Andrieux, Geoffroy, Volkl, Simon, Bengsch, Bertram, Lorenz, Myriam Ricarda, Heeg, Maximilian, Rohr, Jan, Jagle, Sabine, Castro, Carla N., Gross, Miriam, Warthorst, Ursula, Koenig, Christoph, Fuchs, Ilka, Speckmann, Carsten, Thalhammer, Julian, Kapp, Friedrich G., Seidel, Markus G., Duckers, Gregor, Schoenberger, Stefan, Schuetz, Catharina, Fuhrer, Marita, Kobbe, Robin, Holzinger, Dirk, Klemann, Christian, Smisek, Petr, Owens, Stephen, Horneff, Gerd, Kolb, Reinhard, Naumann-Bartsch, Nora, Miano, Maurizio, Staniek, Julian, Rizzi, Marta, Kalina, Tomas, Schneider, Pascal, Erxleben, Anika, Backofen, Rolf, Ekici, Arif, Niemeyer, Charlotte M., Warnatz, Klaus, Grimbacher, Bodo, Eibel, Hermann, Mackensen, Andreas, Frei, Andreas Philipp, Schwarz, Klaus, Boerries, Melanie, Ehl, Stephan, and Rensing-Ehl, Anne

Abstract

The identification and characterization of rare immune cell populations in humans can be facilitated by their growth advantage in the context of specific genetic diseases. Here, we use autoimmune lymphoproliferative syndrome to identify a population of FAS-controlled TCR alpha beta(+) T cells. They include CD4(+), CD8(+), and double-negative T cells and can be defined by a CD38(+)CD45RA(+)T-BET- expression pattern. These unconventional T cells are present in healthy individuals, are generated before birth, are enriched in lymphoid tissue, and do not expand during acute viral infection. They are characterized by a unique molecular signature that is unambiguously different from other known T cell differentiation subsets and independent of CD4 or CD8 expression. Functionally, FAS-controlled T cells represent highly proliferative, noncytotoxic T cells with an IL-10 cytokine bias. Mechanistically, regulation of this physiological population is mediated by FAS and CTLA4 signaling, and its survival is enhanced by mTOR and STAT3 signals. Genetic alterations in these pathways result in expansion of FAS-controlled T cells, which can cause significant lymphoproliferative disease.

Published
2021

181. Subcutaneous dosing regimens of tocilizumab in children with systemic or polyarticular juvenile idiopathic arthritis

Author

Ruperto, Nicolino, Brunner, Hermine, I, Ramanan, Athimalaipet, V, Horneff, Gerd, Cuttica, Ruben, Henrickson, Michael, Anton, Jordi, Lucica Boteanu, Alina, Calvo Penades, Inmaculada, Minden, Kirsten, Schmeling, Heinrike, Hufnagel, Markus, Weiss, Jennifer E., Pardeo, Manuela, Nanda, Kabita, Roth, Johannes, Rubio-Perez, Nadina, Hsu, Joy C., Wimalasundera, Sunethra, Wells, Chris, Bharucha, Kamal, Douglass, Wendy, Bao, Min, Mallalieu, Navita L., Martini, Alberto, Lovell, Daniel, De Benedetti, Fabrizio, Ruperto, Nicolino, Brunner, Hermine, I, Ramanan, Athimalaipet, V, Horneff, Gerd, Cuttica, Ruben, Henrickson, Michael, Anton, Jordi, Lucica Boteanu, Alina, Calvo Penades, Inmaculada, Minden, Kirsten, Schmeling, Heinrike, Hufnagel, Markus, Weiss, Jennifer E., Pardeo, Manuela, Nanda, Kabita, Roth, Johannes, Rubio-Perez, Nadina, Hsu, Joy C., Wimalasundera, Sunethra, Wells, Chris, Bharucha, Kamal, Douglass, Wendy, Bao, Min, Mallalieu, Navita L., Martini, Alberto, Lovell, Daniel, and De Benedetti, Fabrizio

Abstract

Objectives. To determine s.c. tocilizumab (s.c.-TCZ) dosing regimens for systemic JIA (sJIA) and polyarticular JIA (pJIA). Methods. In two 52-week phase 1 b trials, s.c.-TCZ (162 mg/dose) was administered to sJIA patients every week or every 2 weeks (every 10 days before interim analysis) and to pJIA patients every 2 weeks or every 3 weeks with body weight >= 30 kg or <30 kg, respectively. Primary end points were pharmacokinetics, pharmacodynamics and safety; efficacy was exploratory. Comparisons were made to data from phase 3 trials with i.v. tocilizumab (i.v.-TCZ) in sJIA and pJIA. Results. Study participants were 51 sJIA patients and 52 pJIA patients aged 1-17 years who received s.c.-TCZ. Steady-state minimum TCZ concentration (C-trough) >5th percentile of that achieved with i.v.-TCZ was achieved by 49 (96%) sJIA and 52 (100%) pJIA patients. In both populations, pharmacodynamic markers of disease were similar between body weight groups. Improvements in Juvenile Arthritis DAS-71 were comparable between s.c.-TCZ and i.v.-TCZ. By week 52, 53% of sJIA patients and 31% of pJIA patients achieved clinical remission on treatment. Safety was consistent with that of i.v.-TCZ except for injection site reactions, reported by 41.2% and 28.8% of sJIA and pJIA patients, respectively. Infections were reported in 78.4% and 69.2% of patients, respectively. Two sJIA patients died; both deaths were considered to be related to TCZ. Conclusion. s.c.-TCZ provides exposure and risk/benefit profiles similar to those of i.v.-TCZ. S.c. administration provides an alternative administration route that is more convenient for patients and caregivers and that has potential for in-home use.

Published
2021

182. Clinical manifestations and outcome of SARS-CoV-2 infections in children and adolescents with rheumatic musculoskeletal diseases: data from the National Paediatric Rheumatology Database in Germany

Author

Sengler, Claudia, Eulert, Sascha, Minden, Kirsten, Niewerth, Martina, Horneff, Gerd, Kuemmerle-Deschner, Jasmin, Siemer, Caroline, Berendes, Rainer, Girschick, Hermann, Huehn, Regina, Borte, Michael, Hospach, Anton, Emminger, Wolfgang, Armann, Jakob, Klein, Ariane, Kallinich, Tilmann, Sengler, Claudia, Eulert, Sascha, Minden, Kirsten, Niewerth, Martina, Horneff, Gerd, Kuemmerle-Deschner, Jasmin, Siemer, Caroline, Berendes, Rainer, Girschick, Hermann, Huehn, Regina, Borte, Michael, Hospach, Anton, Emminger, Wolfgang, Armann, Jakob, Klein, Ariane, and Kallinich, Tilmann

Abstract

Objectives This study aimed to investigate the clinical manifestations, course and outcome of SARS-CoV-2 infection among children and adolescents with rheumatic and musculoskeletal diseases (RMD). Due to their underlying disease as well due to therapeutic immunosuppression, these patients may be at risk for a severe course of COVID-19 or for a flare of the underlying disease triggered by SARS-CoV-2 infection. Methods Demographic, clinical and treatment data from juvenile patients with RMD as well as data about SARS-CoV-2 infection like test date and method, clinical characteristics, disease course, outcome and impact on the disease activity of the RMD were documented on a specific SARS-CoV-2 questionnaire implemented in the National Paediatric Rheumatology Database (NPRD) in Germany. The survey data were analysed descriptively. Results From 17 April 2020 to 16 February 2021, data were collected from 76 patients (52% female) with RMD and laboratory-proven SARS-CoV-2 infection with median age of 14 years, diagnosed with juvenile idiopathic arthritis (58%), autoinflammatory (24%) and connective tissue disease (8%). Fifty-eight patients (76%) received disease-modifying antirheumatic drugs (DMARDs), 41% biological DMARDs and 11% systemic glucocorticoids. Fifty-eight (76%) had symptoms of COVID-19. Disease course of SARS-CoV-2 infection (classified as asymptomatic, mild, moderate, severe, life-threatening) was mild and outcome of COVID-19 (classified as recovered, not yet recovered, permanent damage or deceased) was good (recovered) in the majority of patients. Two patients were hospitalised, one of whom required intensive care and died of cardiorespiratory failure. In 84% of SARS-CoV-2-positive patients, no relevant increase in disease activity of the RMD was observed. Conclusions In our cohort, SARS-CoV-2 infection in juvenile patients with RMD under various medications was mild with good outcome in the majority of cases and does not appear to have a relevant impact on

Published
2021

183. Long-term follow up of carbohydrate metabolism and adverse events after termination of Omnitrope (R) treatment in children born small for gestational age

Author

Walczak, Mieczyslaw, Szalecki, Mieczyslaw, Horneff, Gerd, Lebl, Jan, Kalina-Faska, Barbara, Giemza, Tomasz, Moldovanu, Florentina, Nanu, Michaela, Zouater, Hichem, Walczak, Mieczyslaw, Szalecki, Mieczyslaw, Horneff, Gerd, Lebl, Jan, Kalina-Faska, Barbara, Giemza, Tomasz, Moldovanu, Florentina, Nanu, Michaela, and Zouater, Hichem

Abstract

Background: Recombinant human growth hormone (rhGH) therapy can affect carbohydrate metabolism and lead to impaired glucose tolerance during treatment. In addition, short children born small for gestational age (SGA) are predisposed to metabolic abnormalities. This study assessed the long-term safety of rhGH (Omnitrope (R)) use in short children born SGA. Methods: This was a follow-up observational study of patients from a phase IV study. The baseline visit was the final visit of the phase IV study. Further visits were planned after 6months (F1), 1year (F2), 5years (F3), and 10years (F4). The primary objective was to evaluate the long-term effect of rhGH treatment on the development of diabetes mellitus; secondary objectives included incidence/severity of adverse events (AEs). Results: In total, 130 subjects were enrolled in the follow-up study; 99 completed F1, 88 completed F2, and 13 completed F3 (no subject reached F4). The full analysis set for evaluation comprised 118 patients (64 female). Mean (standard deviation) duration of follow up was 39.6 (24.4) months. No subject was newly diagnosed with diabetes. The results for carbohydrate metabolism parameters were consistent with this finding. A total of 144 AEs were reported in 54 subjects; these were mostly of mild-to-moderate intensity (96.5%) and not suspected to be related to previous rhGH treatment (94.4%). Serious AEs (n=18) were reported in eight patients; three (in one patient) were suspected as possibly related to previous rhGH treatment (anemia, menorrhagia, oligomenorrhoea). One fatal event occurred (sepsis), which was judged as not related to previous rhGH treatment. Conclusions: None of the participating subjects, who had all been previously treated with Omnitrope (R) in a phase IV study, developed diabetes during this follow-up study. In addition, no other unexpected or concerning safety signals were observed.

Published
2021

184. Efficacy and Safety of Etanercept Biosimilars Compared With the Originator for Treatment of Juvenile Arthritis: A Prospective Observational Study

Author

Thiele, Franz, Klein, Ariane, Hospach, Anton, Windschall, Daniel, Mrusek, Sonja, Ruehlmann, J. Michael, Horneff, Gerd, Thiele, Franz, Klein, Ariane, Hospach, Anton, Windschall, Daniel, Mrusek, Sonja, Ruehlmann, J. Michael, and Horneff, Gerd

Abstract

Objective Analysis of etanercept biosimilars in pediatric patients with juvenile idiopathic arthritis (JIA) in comparison with the etanercept originator in terms of efficacy and safety. Methods Patients diagnosed with JIA who started treatment with either the etanercept originator or a biosimilar after January 1, 2017, were selected from the German BIKER registry (Biologics in Paediatric Rheumatology Registry). Furthermore, patients who started therapy with the originator and switched to a biosimilar during the course of therapy were identified. For both patient groups, disease activity and safety were examined and compared separately. Results After January 1, 2017, 348 patients started treatment with the etanercept originator (n = 293) or a biosimilar (n = 55). Another 57 patients switched to a biosimilar during the course of therapy. A significant decrease or a stable remission of disease activity was observed in both patient groups. The safety profiles were comparable, and frequencies and types of adverse events (AEs) and serious AEs were similar in patients starting therapy with the originator or a biosimilar. Only injection site reactions occurred slightly more frequently under biosimilar therapy, without having an impact on therapy adherence. In patients who switched therapy, the AE rate per 100 patient-years was comparable before (26.4) and after (32.1) the switch. Conclusion In patients with JIA who require treatment with etanercept, the originator is still used much more frequently. However, our study highlights the equivalence of etanercept biosimilars for therapy for JIA. Increased use of these biosimilars in pediatric patients can therefore be recommended without hesitation.

Published
2021

185. Autoinflammation-A clinical and genetic challenge

Author

Horneff, Gerd, Schutz, Catharina, Rosen-Wolff, Angela, Horneff, Gerd, Schutz, Catharina, and Rosen-Wolff, Angela

Abstract

In the last two decades clinical rheumatological practice has been confronted with a steadily increasing number of autoinflammatory diseases, the immunological pathomechanisms of which have been elucidated and in part can be clinically well classified. Whereas targeted genetic diagnostics previously served to confirm a clinically suspected diagnosis, genetic sequencing technology has much improved and enables a new diagnostic approach via high-throughput sequencing, e.g., panel sequencing, whole exome and whole genome sequencing. Thus, the decision to make a diagnosis clinically and/or genetically, has become a daily challenge. This article contrasts the clinical, immunological and genetic aspects of autoinflammatory diseases.

Published
2021

187. Subcutaneous Abatacept in Patients With Polyarticular‐Course Juvenile Idiopathic Arthritis

Author

Brunner, Hermine I., Tzaribachev, Nikolay, Vega‐Cornejo, Gabriel, Louw, Ingrid, Berman, Alberto, Calvo Penadés, Inmaculada, Antón, Jordi, Ávila‐Zapata, Francisco, Cuttica, Rubén, Horneff, Gerd, Foeldvari, Ivan, Keltsev, Vladimir, Kingsbury, Daniel J., Viola, Diego Oscar, Joos, Rik, Lauwerys, Bernard, Paz Gastañaga, Maria Eliana, Rama, Maria Elena, Wouters, Carine, Bohnsack, John, Breedt, Johannes, Fischbach, Michel, Lutz, Thomas, Minden, Kirsten, Miraval, Tatiana, Ally, Mahmood M. T. M., Rubio‐Pérez, Nadina, Solau Gervais, Elisabeth, van Zyl, Riana, Li, Xiaohui, Nys, Marleen, Wong, Robert, Banerjee, Subhashis, Lovell, Daniel J., Martini, Alberto, and Ruperto, Nicolino

Subjects
musculoskeletal diseases, Male, Adolescent, Injections, Subcutaneous, Pediatric Rheumatology, Arthritis, Juvenile, Abatacept, Cohort Studies, Treatment Outcome, Child, Preschool, Humans, Original Article, Female, Child, Immunosuppressive Agents
Abstract

Objective To investigate the pharmacokinetics, effectiveness, and safety of subcutaneous (SC) abatacept treatment over 24 months in patients with polyarticular‐course juvenile idiopathic arthritis (JIA). Methods In this phase III, open‐label, international, multicenter, single‐arm study, patients with polyarticular JIA (cohort 1, ages 6–17 years and cohort 2, ages 2–5 years) in whom treatment with ≥1 disease‐modifying antirheumatic drug was unsuccessful received weight‐tiered SC abatacept weekly: 10 to

Published
2018

189. The NEMO mutation creating the most-upstream premature stop codon is hypomorphic because of a reinitiation of translation

Author

Puel, Anne, Reichenbach, Janine, Bustamante, Jacinta, Ku, Cheng-Lung, Feinberg, Jacqueline, Doffinger, Rainer, Bonnet, Marion, Filipe-Santos, Orchidee, de Beaucoudrey, Ludovic, Durandy, Anne, Horneff, Gerd, Novelli, Francesco, Wahn, Volker, Smahi, Asma, Israel, Alain, Niehues, Tim, and Casanova, Jean-Laurent

Subjects
Codon -- Research, Gene mutations -- Research, Biological sciences
Published
2006

193. Leflunomide or methotrexate for juvenile rheumatoid arthritis

Author

Silverman, Earl, Spiegel, Lynn, Saurenmann, Rotraud K., Horneff, Gerd, Szer, Ilona S., Mouy, Richard, Jung, Lawrence K., Lahdenne, Pekka, Calvo, Immaculada, Simpson, Karen, Stewart, John A., and Strand, Vibeke

Subjects
Methotrexate -- Comparative analysis, Methotrexate -- Complications and side effects, Leflunomide -- Comparative analysis, Leflunomide -- Complications and side effects, Rheumatoid arthritis in children -- Care and treatment
Abstract

The safety and efficacy of leflunomide was compared with that of methotrexate in the treatment of polyarticular juvenile rheumatoid arthritis in a multinational, randomized, controlled trial. It was concluded that in patients with polyarticular juvenile rheumatoid arthritis, methotrexate and leflunomide both resulted in high rates of clinical improvement with a slight greater rate for methotrexate.

Published
2005

194. Functional flow cytometry of monocytes for routine diagnosis of innate primary immunodeficiencies

Author

Ammann, Sandra, Fuchs, Sebastian, Martin-Martin, Lydia, Castro, Carla N., Spielberger, Benedikt, Klemann, Christian, Elling, Roland, Heeg, Maximilian, Speckmann, Carsten, Hainmann, Ina, Kaiser-Labusch, Petra, Horneff, Gerd, Thalhammer, Julian, Bredius, Robbert G., Stadt, Udo zur, Lehmberg, Kai, Fuchs, Ilka, von Spee-Mayer, Caroline, Henneke, Philipp, and Ehl, Stephan

Published
2020
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196. MRP8/14 serum levels as diagnostic markers for systemic juvenile idiopathic arthritis in children with prolonged fever.

Author

Park, Carolin, Miranda-Garcia, María, Berendes, Rainer, Horneff, Gerd, Kuemmerle-Deschner, Jasmin, Ganser, Gerd, Huppertz, Hans-Iko, Minden, Kirsten, Haas, Johannes-Peter, Jansson, Annette F, Borte, Michael, Schuetz, Catharina, Oommen, Prasad, Frosch, Michael, Schlueter, Bernhard, Richter-Unruh, Annette, Kessel, Christoph, Hinze, Claas, Wittkowski, Helmut, and Roth, Johannes

Subjects
BIOMARKERS, INTERLEUKINS, POINT-of-care testing, FERRITIN, ANTI-inflammatory agents, SERUM, JUVENILE idiopathic arthritis, DIFFERENTIAL diagnosis, RETROSPECTIVE studies, CALCITONIN, IMMUNOASSAY, INFECTION, ENZYME-linked immunosorbent assay, BLOOD sedimentation, RHEUMATIC fever in children, SENSITIVITY & specificity (Statistics)
Abstract

Objectives Differential diagnosis in children with prolonged fever is challenging. In particular, differentiating systemic-onset JIA (SJIA) from infectious diseases is difficult. Biomarkers are needed that support the diagnostic work-up. The aim of this study was to validate the usefulness of Myeloid-related protein 8/14 (MRP8/14) measurements in the diagnostic work-up of febrile children and to transfer it to clinical practice. Methods Data for 1110 paediatric patients were included and divided into two cohorts: (cohort A) for validation of MRP8/14 test performance with three different testing systems: the experimental ELISA, commercial ELISA and an innovative (point-of-care test) lateral flow immunoassay (LFIA); (cohort B) to validate the diagnostic accuracy with the two latter assays. Results In cohort A (n  = 940), MRP8/14 was elevated in SJIA (12 110 ± 2650 ng/ml mean ± 95% CI) compared with other diagnoses (including infections and autoinflammatory diseases; 2980 ± 510 ng/ml) irrespective of fever and anti-inflammatory treatment (P  < 0.001). In untreated patients with fever (n  = 195) MRP8/14 levels in SJIA (19 740 ± 5080 ng/ml) were even higher compared with other diagnoses (4590 ± 1160 ng/ml) (P  < 0.001, sensitivity 73%, specificity 90%). In group B1, the performance of the tests was confirmed in untreated patients with fever (n  = 170): commercial ELISA (sensitivity 79%, specificity 89%) and LFIA (sensitivity 84%, specificity 81%). Compared with ferritin, IL-18, ESR, soluble IL-2 receptor and procalcitonin, MRP8/14 showed the best accuracy. Conclusion MRP8/14 serum analyses have been validated as a helpful tool supporting the diagnosis of SJIA in febrile children. The results could be confirmed with commercial ELISA and LFIA enabling a rapid diagnostic point-of-care screening test. [ABSTRACT FROM AUTHOR]

Published
2022
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197. Two Randomized Trials of Canakinumab in Systemic Juvenile Idiopathic Arthritis

Author

Ruperto, Nicolino, Brunner, Hermine I., Quartier, Pierre, Constantin, Tamás, Wulffraat, Nico, Horneff, Gerd, Brik, Riva, McCann, Liza, Kasapcopur, Ozgur, Rutkowska-Sak, Lidia, Schneider, Rayfel, Berkun, Yackov, Calvo, Inmaculada, Erguven, Muferet, Goffin, Laurence, Hofer, Michael, Kallinich, Tilmann, Oliveira, Sheila K., Uziel, Yosef, Viola, Stefania, Nistala, Kiran, Wouters, Carine, Cimaz, Rolando, Ferrandiz, Manuel A., Flato, Berit, Gamir, Maria Luz, Kone-Paut, Isabelle, Grom, Alexei, Magnusson, Bo, Ozen, Seza, Sztajnbok, Flavio, Lheritier, Karine, Abrams, Ken, Kim, Dennis, Martini, Alberto, and Lovell, Daniel J.

Published
2012
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198. Efficacy and Safety of Tocilizumab in Patients with Polyarticular Juvenile Idiopathic Arthritis: Data From a Phase 3 Trial.: 1597

Author

Brunner, Hermine, Ruperto, Nicolino, Zuber, Zbigniew, Keane, Caroline, Harari, Olivier, Kenwright, Andrew, Cuttica, Ruben J., Keltsev, Vladimir, Xavier, Ricardo, Penades, Inmaculada Calvo, Nikishina, Irina, Rubio-Perez, Nadina, Alekseeva, Ekaterina, Chasnyk, Vyacheslav, Chavez, Jose, Horneff, Gerd, Opoka-Winiarska, Violetta, Quartier, Pierre, Silva, Clovis A., Silverman, Earl D., Spindler, Alberto, Lovell, D. J., Martini, Alberto, and De Benedetti, Fabrizio

Published
2012

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