Wu, Wanlong, Jordan, Suzana ORCID: 0000-0002-8114-8239, Graf, Nicole, Pena, Janethe de Oliveira, Curram, John, Allanore, Yannick, Matucci-Cerinic, Marco, Pope, Janet E., Denton, Christopher P., Khanna, Dinesh, Distler, Oliver ORCID: 0000-0002-0546-8310, Matucci-Cerinic, Marco, Guiducci, Serena ORCID: 0000-0003-2722-6475, Walker, Ulrich, Jaeger, Veronika ORCID: 0000-0002-6913-0976, Bannert, Bettina, Lapadula, Giovanni ORCID: 0000-0002-8427-6092, Becvarare, Radim, Cutolo, Maurizio, Valentini, Gabriele, Siegert, Elise, Rednic, Simona, Allanore, Yannick, Montecucco, C., Carreira, Patricia E., Novak, Srdan ORCID: 0000-0001-8870-4704, Czirjak, Laszlo, Varju, Cecilia ORCID: 0000-0002-6849-9114, Chizzolini, Carlo, Allai, Daniela, Kucharz, Eugene J., Cozzi, Franco, Rozman, Blaz, Mallia, Carmel, Gabrielli, Armando, Bancel, Dominique Farge, Airo, Paolo, Hesselstrand, Roger, Martinovic, Duska, Balbir-Gurman, Alexandra, Braun-Moscovici, Yolanda, Hunzelmann, Nicolas, Pellerito, Raffaele, Caramaschi, Paola, Black, Carol, Damjanov, Nemanja, Henes, Joerg, Ortiz Santamaria, Vera, Heitmann, Stefan, Seidel, Matthias, Pereira Da Silva, Jose Antonio, Stamenkovic, Bojana, Selmi, Carlo Francesco, Tikly, Mohammed, Denisov, Lev N., Mueller-Ladner, Ulf, Engelhart, Merete, Hachulla, Eric, Riccieri, Valeria, Ionescu, Ruxandra Maria, Mihai, Carina, Sunderkoetter, Cord, Kuhn, Annegret, Schett, Georg, Distler, Joerg, Meroni, Pierluigi, Ingegnoli, Francesca, Mouthon, Luc, De Keyser, Filip, Smith, Vanessa, Cantatore, Francesco Paolo, Corrado, Ada, Ullman, Susanne, Iversen, Line, Pozzi, Maria Rosa, Eyerich, Kilian, Hein, Ruediger, Knott, Elisabeth, Wiland, Piotr, Szmyrka-Kaczmarek, Magdalena, Sokolik, Renata, Morgiel, Ewa, Madej, Marta, Jose Alegre-Sancho, Juan, Krummel-Lorenz, Brigitte, Saar, Petra, Aringer, Martin, Guenther, Claudia, Anne, Erler, Westhovens, Rene, De Langhe, Ellen, Lenaerts, Jan, Anic, Branimir ORCID: 0000-0003-4940-0577, Baresic, Marko, Mayer, Miroslav, Uprus, Maria, Otsa, Kati, Yavuz, Sule, Radominski, Sebastiao Cezar, Mueller, Carolina de Souza, Azevedo, Valderilio Feijo, Popa, Sergei, Zenone, Thierry, Stebbings, Simon, Highton, John, Mathieu, Alessandro, Vacca, Alessandra, Stamp, Lisa, Chapman, Peter, O'Donnell, John, Solanki, Kamal, Doube, Alan, Veale, Douglas ORCID: 0000-0003-2802-4971, O'Rourke, Marie, Loyo, Esthela, Li, Mengtao, Rosato, Edoardo ORCID: 0000-0002-7417-8093, Amoroso, Antonio, Gigante, Antonietta, Oksel, Fahrettin, Yargucu, Figen, Tanaseanu, Cristina-Mihaela, Popescu, Monica, Dumitrascu, Alina, Tiglea, Isabela, Foti, Rosario ORCID: 0000-0002-7301-8362, Visalli, Elisa, Benenati, Alessia, Amato, Giorgio, Ancuta, Codrina, Chirieac, Rodica, Villiger, Peter, Adler, Sabine, Dan, Diana, de la Pena Lefebvre, Paloma Garcia, Rodriguez Rubio, Silvia, Valero Exposito, Marta, Sibilia, Jean, Chatelus, Emmanuel, Gottenberg, Jacques Eric, Chifflot, Helene, Litinsky, Ira, Del Galdo, Francesco, Venalis, Algirdas, Saketkoo, Lesley Ann, Lasky, Joseph A., Kerzberg, Eduardo, Montoya, Fabiana, Cosentino, Vanesa, Limonta, Massimiliano, Brucato, Antonio Luca, Lupi, Elide, Spertini, Francois, Ribi, Camillo, Buss, Guillaume, Martin, Thierry, Guffroy, Aurelien, Poindron, Vincent, Chung, Lori, Schmeiser, Tim, Zebryk, Pawel, Riso, Nuno, Riemekasten, Gabriela, Rezus, Elena ORCID: 0000-0002-8175-2583 and Puttini, Piercarlo Sarzi (2019). Progressive skin fibrosis is associated with a decline in lung function and worse survival in patients with diffuse cutaneous systemic sclerosis in the European Scleroderma Trials and Research (EUSTAR) cohort. Ann. Rheum. Dis., 78 (5). S. 648 - 657. LONDON: BMJ PUBLISHING GROUP. ISSN 1468-2060

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Abstract

Objectives To determine whether progressive skin fibrosis is associated with visceral organ progression and mortality during follow-up in patients with diffuse cutaneous systemic sclerosis (dcSSc). Methods We evaluated patients from the European Scleroderma Trials and Research database with dcSSc, baseline modified Rodnan skin score (mRSS) >= 7, valid mRSS at 12 +/- 3 months after baseline and >= 1 annual follow-up visit. Progressive skin fibrosis was defined as an increase in mRSS >5 and >= 25% from baseline to 12 +/- 3 months. Outcomes were pulmonary, cardiovascular and renal progression, and all-cause death. Associations between skin progression and outcomes were evaluated by Kaplan-Meier survival analysis and multivariable Cox regression. Results Of 1021 included patients, 78 (7.6%) had progressive skin fibrosis (skin progressors). Median follow-up was 3.4 years. Survival analyses indicated that skin progressors had a significantly higher probability of FVC decline >= 10% (53.6% vs 34.4%; p<0.001) and all-cause death (15.4% vs 7.3%; p=0.003) than non-progressors. These significant associations were also found in subgroup analyses of patients with either low baseline mRSS (<= 22/51) or short disease duration (<= 15 months). In multivariable analyses, skin progression within 1 year was independently associated with FVC decline >= 10% (HR 1.79, 95% CI 1.20 to 2.65) and all-cause death (HR 2.58, 95% CI 1.31 to 5.09). Conclusions Progressive skin fibrosis within 1 year is associated with decline in lung function and worse survival in dcSSc during follow-up. These results confirm mRSS as a surrogate marker in dcSSc, which will be helpful for cohort enrichment in future trials and risk stratification in clinical practice.

Item Type: Journal Article
Creators:
CreatorsEmailORCIDORCID Put Code
Wu, WanlongUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jordan, SuzanaUNSPECIFIEDorcid.org/0000-0002-8114-8239UNSPECIFIED
Graf, NicoleUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pena, Janethe de OliveiraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Curram, JohnUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Allanore, YannickUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Matucci-Cerinic, MarcoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pope, Janet E.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Denton, Christopher P.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Khanna, DineshUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, OliverUNSPECIFIEDorcid.org/0000-0002-0546-8310UNSPECIFIED
Matucci-Cerinic, MarcoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Guiducci, SerenaUNSPECIFIEDorcid.org/0000-0003-2722-6475UNSPECIFIED
Walker, UlrichUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jaeger, VeronikaUNSPECIFIEDorcid.org/0000-0002-6913-0976UNSPECIFIED
Bannert, BettinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lapadula, GiovanniUNSPECIFIEDorcid.org/0000-0002-8427-6092UNSPECIFIED
Becvarare, RadimUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Cutolo, MaurizioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Valentini, GabrieleUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Siegert, EliseUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rednic, SimonaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Allanore, YannickUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Montecucco, C.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Carreira, Patricia E.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Novak, SrdanUNSPECIFIEDorcid.org/0000-0001-8870-4704UNSPECIFIED
Czirjak, LaszloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Varju, CeciliaUNSPECIFIEDorcid.org/0000-0002-6849-9114UNSPECIFIED
Chizzolini, CarloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Allai, DanielaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kucharz, Eugene J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Cozzi, FrancoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rozman, BlazUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mallia, CarmelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gabrielli, ArmandoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Bancel, Dominique FargeUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Airo, PaoloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hesselstrand, RogerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Martinovic, DuskaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Balbir-Gurman, AlexandraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Braun-Moscovici, YolandaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hunzelmann, NicolasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pellerito, RaffaeleUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Caramaschi, PaolaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Black, CarolUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Damjanov, NemanjaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Henes, JoergUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ortiz Santamaria, VeraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Heitmann, StefanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Seidel, MatthiasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pereira Da Silva, Jose AntonioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stamenkovic, BojanaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Selmi, Carlo FrancescoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Tikly, MohammedUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Denisov, Lev N.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mueller-Ladner, UlfUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Engelhart, MereteUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hachulla, EricUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riccieri, ValeriaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ionescu, Ruxandra MariaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mihai, CarinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Sunderkoetter, CordUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kuhn, AnnegretUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Schett, GeorgUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, JoergUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Meroni, PierluigiUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ingegnoli, FrancescaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mouthon, LucUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
De Keyser, FilipUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Smith, VanessaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Cantatore, Francesco PaoloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Corrado, AdaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ullman, SusanneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Iversen, LineUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pozzi, Maria RosaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Eyerich, KilianUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hein, RuedigerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Knott, ElisabethUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Wiland, PiotrUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Szmyrka-Kaczmarek, MagdalenaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Sokolik, RenataUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Morgiel, EwaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Madej, MartaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jose Alegre-Sancho, JuanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Krummel-Lorenz, BrigitteUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Saar, PetraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Aringer, MartinUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Guenther, ClaudiaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Anne, ErlerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Westhovens, ReneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
De Langhe, EllenUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lenaerts, JanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Anic, BranimirUNSPECIFIEDorcid.org/0000-0003-4940-0577UNSPECIFIED
Baresic, MarkoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mayer, MiroslavUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Uprus, MariaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Otsa, KatiUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Yavuz, SuleUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Radominski, Sebastiao CezarUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mueller, Carolina de SouzaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Azevedo, Valderilio FeijoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Popa, SergeiUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Zenone, ThierryUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stebbings, SimonUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Highton, JohnUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mathieu, AlessandroUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Vacca, AlessandraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stamp, LisaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chapman, PeterUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
O'Donnell, JohnUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Solanki, KamalUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Doube, AlanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Veale, DouglasUNSPECIFIEDorcid.org/0000-0003-2802-4971UNSPECIFIED
O'Rourke, MarieUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Loyo, EsthelaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Li, MengtaoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rosato, EdoardoUNSPECIFIEDorcid.org/0000-0002-7417-8093UNSPECIFIED
Amoroso, AntonioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gigante, AntoniettaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Oksel, FahrettinUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Yargucu, FigenUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Tanaseanu, Cristina-MihaelaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Popescu, MonicaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Dumitrascu, AlinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Tiglea, IsabelaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Foti, RosarioUNSPECIFIEDorcid.org/0000-0002-7301-8362UNSPECIFIED
Visalli, ElisaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Benenati, AlessiaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Amato, GiorgioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ancuta, CodrinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chirieac, RodicaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Villiger, PeterUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Adler, SabineUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Dan, DianaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
de la Pena Lefebvre, Paloma GarciaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rodriguez Rubio, SilviaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Valero Exposito, MartaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Sibilia, JeanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chatelus, EmmanuelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gottenberg, Jacques EricUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chifflot, HeleneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Litinsky, IraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Del Galdo, FrancescoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Venalis, AlgirdasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Saketkoo, Lesley AnnUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lasky, Joseph A.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kerzberg, EduardoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Montoya, FabianaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Cosentino, VanesaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Limonta, MassimilianoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Brucato, Antonio LucaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lupi, ElideUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Spertini, FrancoisUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ribi, CamilloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Buss, GuillaumeUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Martin, ThierryUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Guffroy, AurelienUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Poindron, VincentUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chung, LoriUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Schmeiser, TimUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Zebryk, PawelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riso, NunoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riemekasten, GabrielaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rezus, ElenaUNSPECIFIEDorcid.org/0000-0002-8175-2583UNSPECIFIED
Puttini, Piercarlo SarziUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
URN: urn:nbn:de:hbz:38-149095
DOI: 10.1136/annrheumdis-2018-213455
Journal or Publication Title: Ann. Rheum. Dis.
Volume: 78
Number: 5
Page Range: S. 648 - 657
Date: 2019
Publisher: BMJ PUBLISHING GROUP
Place of Publication: LONDON
ISSN: 1468-2060
Language: English
Faculty: Unspecified
Divisions: Unspecified
Subjects: no entry
Uncontrolled Keywords:
KeywordsLanguage
THICKNESS SCORE; PROGNOSTIC-FACTORS; PREDICTION; INVOLVEMENT; MORTALITY; DEATH; VARIABILITY; IMPROVEMENT; VALIDATIONMultiple languages
RheumatologyMultiple languages
Refereed: Yes
URI: http://kups.ub.uni-koeln.de/id/eprint/14909

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