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Mapping and predicting mortality from systemic sclerosis

Author:
Elhai, Muriel  Meune, Christophe  Boubaya, Marouane  Avouac, Jerome  Hachulla, Eric  Balbir-Gurman, Alexandra  Riemekasten, Gabriela  Airo, Paolo  Joven, Beatriz  Vettori, Serena  Cozzi, Franco  Ullman, Susanne  Czirjak, Laszlo  Tikly, Mohammed  Mueller-Ladner, U. L. F.  Caramaschi, Paola  Distler, Oliver  Iannone, Florenzo  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  Heitmann, Stefan  Distler, Joerg H. W.  Zenone, Thierry  Seidel, Matthias  Vacca, Alessandra  De langhe, Ellen  Novak, Srdan  Cutolo, Maurizio  Mouthon, Luc  Henes, Joerg  Chizzolini, Carlo  von Muhlen, Carlos Alberto  Solanki, Kamal  Rednic, Simona  Stamp, Lisa  Anic, Branimir  Santamaria, Vera Ortiz  De Santis, Maria  Yavuz, Sule  Alberto Sifuentes-Giraldo, Walter  Chatelus, Emmanuel  Stork, Jiri  van Laar, Jacob  Loyo, Esthela  de la Pena Lefebvre, Paloma Garcia  Eyerich, Kilian  Cosentino, Vanesa  Jose Alegre-Sancho, Juan  Kowal-Bielecka, Otylia  Rey, Gregoire  Matucci-Cerinic, Marco  Allanore, Yannick  


Journal:
ANNALS OF THE RHEUMATIC DISEASES


Issue Date:
2017


Abstract(summary):

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.


Page:
1897---1905


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