Elhai, Muriel ORCID: 0000-0001-8627-5758, Meune, Christophe, Boubaya, Marouane, Avouac, Jerome, Hachulla, Eric, Balbir-Gurman, Alexandra, Riemekasten, Gabriela, Airo, Paolo, Joven, Beatriz, Vettori, Serena ORCID: 0000-0001-5617-9829, Cozzi, Franco, Ullman, Susanne, Czirjak, Laszlo, Tikly, Mohammed ORCID: 0000-0001-7850-3538, Mueller-Ladner, U. L. F., Caramaschi, Paola, Distler, Oliver ORCID: 0000-0002-0546-8310, Iannone, Florenzo ORCID: 0000-0003-0474-5344, Ananieva, Lidia P., Hesselstrand, Roger, Becvar, Radim, Gabrielli, Armando, Damjanov, Nemanja, Salvador, Maria J., Riccieri, Valeria, Mihai, Carina, Szucs, Gabriella, Walker, Ulrich A., Hunzelmann, Nicolas, Martinovic, Duska, Smith, Vanessa, Mueller, Carolina de Souza, Montecucco, Carlo Maurizio, Opris, Daniela, Ingegnoli, Francesca, Vlachoyiannopoulos, Panayiotis G., Stamenkovic, Bojana, Rosato, Edoardo ORCID: 0000-0002-7417-8093, Heitmann, Stefan, Distler, Joerg H. W., Zenone, Thierry, Seidel, Matthias, Vacca, Alessandra, De langhe, Ellen, Novak, Srdan ORCID: 0000-0001-8870-4704, Cutolo, Maurizio, Mouthon, Luc, Henes, Joerg, Chizzolini, Carlo, von Muhlen, Carlos Alberto, Solanki, Kamal, Rednic, Simona, Stamp, Lisa, Anic, Branimir ORCID: 0000-0003-4940-0577, Santamaria, Vera Ortiz, De Santis, Maria, Yavuz, Sule, Alberto Sifuentes-Giraldo, Walter, Chatelus, Emmanuel, Stork, Jiri ORCID: 0000-0002-8505-3560, van Laar, Jacob, Loyo, Esthela, de la Pena Lefebvre, Paloma Garcia, Eyerich, Kilian, Cosentino, Vanesa, Jose Alegre-Sancho, Juan, Kowal-Bielecka, Otylia ORCID: 0000-0002-0613-1306, Rey, Gregoire ORCID: 0000-0001-7291-9444, Matucci-Cerinic, Marco and Allanore, Yannick (2017). Mapping and predicting mortality from systemic sclerosis. Ann. Rheum. Dis., 76 (11). S. 1897 - 1906. LONDON: BMJ PUBLISHING GROUP. ISSN 1468-2060

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Abstract

Objectives To determine the causes of death and risk factors in systemic sclerosis (SSc). Methods Between 2000 and 2011, we examined the death certificates of all French patients with SSc to determine causes of death. Then we examined causes of death and developed a score associated with all-cause mortality from the international European Scleroderma Trials and Research (EUSTAR) database. Candidate prognostic factors were tested by Cox proportional hazards regression model by single variable analysis, followed by a multiple variable model stratified by centres. The bootstrapping technique was used for internal validation. Results We identified 2719 French certificates of deaths related to SSc, mainly from cardiac (31%) and respiratory (18%) causes, and an increase in SSc-specific mortality over time. Over a median follow-up of 2.3 years, 1072 (9.6%) of 11 193 patients from the EUSTAR sample died, from cardiac disease in 27% and respiratory causes in 17%. By multiple variable analysis, a risk score was developed, which accurately predicted the 3-year mortality, with an area under the curve of 0.82. The 3-year survival of patients in the upper quartile was 53%, in contrast with 98% in the first quartile. Conclusion Combining two complementary and detailed databases enabled the collection of an unprecedented 3700 deaths, revealing the major contribution of the cardiopulmonary system to SSc mortality. We also developed a robust score to risk-stratify these patients and estimate their 3-year survival. With the emergence of new therapies, these important observations should help caregivers plan and refine the monitoring and management to prolong these patients' survival.

Item Type: Journal Article
Creators:
CreatorsEmailORCIDORCID Put Code
Elhai, MurielUNSPECIFIEDorcid.org/0000-0001-8627-5758UNSPECIFIED
Meune, ChristopheUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Boubaya, MarouaneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Avouac, JeromeUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hachulla, EricUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Balbir-Gurman, AlexandraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riemekasten, GabrielaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Airo, PaoloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Joven, BeatrizUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Vettori, SerenaUNSPECIFIEDorcid.org/0000-0001-5617-9829UNSPECIFIED
Cozzi, FrancoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ullman, SusanneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Czirjak, LaszloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Tikly, MohammedUNSPECIFIEDorcid.org/0000-0001-7850-3538UNSPECIFIED
Mueller-Ladner, U. L. F.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Caramaschi, PaolaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, OliverUNSPECIFIEDorcid.org/0000-0002-0546-8310UNSPECIFIED
Iannone, FlorenzoUNSPECIFIEDorcid.org/0000-0003-0474-5344UNSPECIFIED
Ananieva, Lidia P.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hesselstrand, RogerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Becvar, RadimUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gabrielli, ArmandoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Damjanov, NemanjaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Salvador, Maria J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riccieri, ValeriaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mihai, CarinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Szucs, GabriellaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Walker, Ulrich A.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hunzelmann, NicolasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Martinovic, DuskaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Smith, VanessaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mueller, Carolina de SouzaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Montecucco, Carlo MaurizioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Opris, DanielaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ingegnoli, FrancescaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Vlachoyiannopoulos, Panayiotis G.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stamenkovic, BojanaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rosato, EdoardoUNSPECIFIEDorcid.org/0000-0002-7417-8093UNSPECIFIED
Heitmann, StefanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, Joerg H. W.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Zenone, ThierryUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Seidel, MatthiasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Vacca, AlessandraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
De langhe, EllenUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Novak, SrdanUNSPECIFIEDorcid.org/0000-0001-8870-4704UNSPECIFIED
Cutolo, MaurizioUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mouthon, LucUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Henes, JoergUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chizzolini, CarloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
von Muhlen, Carlos AlbertoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Solanki, KamalUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rednic, SimonaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stamp, LisaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Anic, BranimirUNSPECIFIEDorcid.org/0000-0003-4940-0577UNSPECIFIED
Santamaria, Vera OrtizUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
De Santis, MariaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Yavuz, SuleUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Alberto Sifuentes-Giraldo, WalterUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chatelus, EmmanuelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stork, JiriUNSPECIFIEDorcid.org/0000-0002-8505-3560UNSPECIFIED
van Laar, JacobUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Loyo, EsthelaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
de la Pena Lefebvre, Paloma GarciaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Eyerich, KilianUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Cosentino, VanesaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jose Alegre-Sancho, JuanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kowal-Bielecka, OtyliaUNSPECIFIEDorcid.org/0000-0002-0613-1306UNSPECIFIED
Rey, GregoireUNSPECIFIEDorcid.org/0000-0001-7291-9444UNSPECIFIED
Matucci-Cerinic, MarcoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Allanore, YannickUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
URN: urn:nbn:de:hbz:38-213265
DOI: 10.1136/annrheumdis-2017-211448
Journal or Publication Title: Ann. Rheum. Dis.
Volume: 76
Number: 11
Page Range: S. 1897 - 1906
Date: 2017
Publisher: BMJ PUBLISHING GROUP
Place of Publication: LONDON
ISSN: 1468-2060
Language: English
Faculty: Unspecified
Divisions: Unspecified
Subjects: no entry
Uncontrolled Keywords:
KeywordsLanguage
EULAR SCLERODERMA TRIALS; EUSTAR DATABASE; 5-YEAR SURVIVAL; COHORT; MODEL; METAANALYSIS; DEATH; POPULATION; VALIDATION; PROFILEMultiple languages
RheumatologyMultiple languages
Refereed: Yes
URI: http://kups.ub.uni-koeln.de/id/eprint/21326

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