Herrick, Ariane L., Peytrignet, Sebastien, Lunt, Mark ORCID: 0000-0002-2391-5575, Pan, Xiaoyan, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan J., Dinsdale, Graham ORCID: 0000-0001-6245-2721, Brown, Edith, Czirjak, Laszlo, Distler, Joerg H. W., Distler, Oliver ORCID: 0000-0002-0546-8310, Fligelstone, Kim, Gregory, William J., Ochiel, Rachel, Vonk, Madelon C., Ancuta, Codrina, Ong, Voon H., Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Oyvind, Jobanputra, Paresh ORCID: 0000-0002-2712-667X, Jordan, Alison C., Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C., Agard, Christian, Anderson, Marina E., Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H., Chung, Lorinda, Damjanov, Nemanja S., Gunawardena, Harsha, Lanyon, Peter ORCID: 0000-0002-9855-6802, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren ORCID: 0000-0002-5654-4993, MacGregor, Alexander J., McHugh, Neil, Mueller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E., Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S., van Laar, Jacob M., Pathare, Sanjay, Proudman, Susanna M., Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim and Denton, Christopher P. (2018). Patterns and predictors of skin score change in early diffuse systemic sclerosis from the European Scleroderma Observational Study. Ann. Rheum. Dis., 77 (4). S. 563 - 571. LONDON: BMJ PUBLISHING GROUP. ISSN 1468-2060

Full text not available from this repository.

Abstract

Objectives Our aim was to use the opportunity provided by the European Scleroderma Observational Study to (1) identify and describe those patients with early diffuse cutaneous systemic sclerosis (dcSSc) with progressive skin thickness, and (2) derive prediction models for progression over 12 months, to inform future randomised controlled trials (RCTs). Methods The modified Rodnan skin score (mRSS) was recorded every 3months in 326 patients. Progressors' were defined as those experiencing a 5-unit and 25% increase in mRSS score over 12 months (3 months). Logistic models were fitted to predict progression and, using receiver operating characteristic (ROC) curves, were compared on the basis of the area under curve (AUC), accuracy and positive predictive value (PPV). Results 66 patients (22.5%) progressed, 227 (77.5%) did not (33 could not have their status assessed due to insufficient data). Progressors had shorter disease duration (median 8.1 vs 12.6 months, P=0.001) and lower mRSS (median 19 vs 21 units, P=0.030) than non-progressors. Skin score was highest, and peaked earliest, in the anti-RNA polymerase III (Pol3+) subgroup (n=50). A first predictive model (including mRSS, duration of skin thickening and their interaction) had an accuracy of 60.9%, AUC of 0.666 and PPV of 33.8%. By adding a variable for Pol3 positivity, the model reached an accuracy of 71%, AUC of 0.711 and PPV of 41%. Conclusions Two prediction models for progressive skin thickening were derived, for use both in clinical practice and for cohort enrichment in RCTs. These models will inform recruitment into the many clinical trials of dcSSc projected for the coming years. Trial registration number NCT02339441.

Item Type: Journal Article
Creators:
CreatorsEmailORCIDORCID Put Code
Herrick, Ariane L.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Peytrignet, SebastienUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lunt, MarkUNSPECIFIEDorcid.org/0000-0002-2391-5575UNSPECIFIED
Pan, XiaoyanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hesselstrand, RogerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mouthon, LucUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Silman, Alan J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Dinsdale, GrahamUNSPECIFIEDorcid.org/0000-0001-6245-2721UNSPECIFIED
Brown, EdithUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Czirjak, LaszloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, Joerg H. W.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Distler, OliverUNSPECIFIEDorcid.org/0000-0002-0546-8310UNSPECIFIED
Fligelstone, KimUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gregory, William J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ochiel, RachelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Vonk, Madelon C.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ancuta, CodrinaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Ong, Voon H.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Farge, DominiqueUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hudson, MarieUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Matucci-Cerinic, MarcoUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Balbir-Gurman, AlexandraUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Midtvedt, OyvindUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jobanputra, PareshUNSPECIFIEDorcid.org/0000-0002-2712-667XUNSPECIFIED
Jordan, Alison C.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Stevens, WendyUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Moinzadeh, PiaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hall, Frances C.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Agard, ChristianUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Anderson, Marina E.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Diot, ElisabethUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Madhok, RajanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Akil, MohammedUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Buch, Maya H.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chung, LorindaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Damjanov, Nemanja S.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gunawardena, HarshaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lanyon, PeterUNSPECIFIEDorcid.org/0000-0002-9855-6802UNSPECIFIED
Ahmad, YasmeenUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Chakravarty, KuntalUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Jacobsen, SorenUNSPECIFIEDorcid.org/0000-0002-5654-4993UNSPECIFIED
MacGregor, Alexander J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
McHugh, NeilUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mueller-Ladner, UlfUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Riemekasten, GabrielaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Becker, MichaelUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Roddy, JanetUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Carreira, Patricia E.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Fauchais, Anne LaureUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hachulla, EricUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hamilton, JenniferUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Inanc, MuratUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
McLaren, John S.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
van Laar, Jacob M.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pathare, SanjayUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Proudman, Susanna M.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rudin, AnnaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Sahhar, JoanneUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Coppere, BrigitteUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Serratrice, ChristineUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Sheeran, TomUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Veale, Douglas J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Grange, ClaireUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Trad, Georges-SelimUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Denton, Christopher P.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
URN: urn:nbn:de:hbz:38-191033
DOI: 10.1136/annrheumdis-2017-211912
Journal or Publication Title: Ann. Rheum. Dis.
Volume: 77
Number: 4
Page Range: S. 563 - 571
Date: 2018
Publisher: BMJ PUBLISHING GROUP
Place of Publication: LONDON
ISSN: 1468-2060
Language: English
Faculty: Unspecified
Divisions: Unspecified
Subjects: no entry
Uncontrolled Keywords:
KeywordsLanguage
DOUBLE-BLIND; THICKNESS SCORE; D-PENICILLAMINE; PLACEBO; MULTICENTER; FIBROSIS; THERAPY; TRIALMultiple languages
RheumatologyMultiple languages
Refereed: Yes
URI: http://kups.ub.uni-koeln.de/id/eprint/19103

Downloads

Downloads per month over past year

Altmetric

Export

Actions (login required)

View Item View Item