Early trajectories of skin thickening are associated with severity and mortality in systemic sclerosis.
Clinical heterogeneity
Modified Rodnan skin score
Skin thickening trajectories
Systemic sclerosis
Journal
Arthritis research & therapy
ISSN: 1478-6362
Titre abrégé: Arthritis Res Ther
Pays: England
ID NLM: 101154438
Informations de publication
Date de publication:
18 02 2020
18 02 2020
Historique:
received:
08
10
2019
accepted:
31
01
2020
entrez:
20
2
2020
pubmed:
20
2
2020
medline:
25
11
2020
Statut:
epublish
Résumé
Systemic sclerosis (SSc) is a severe and highly heterogeneous disease. The modified Rodnan skin score (mRSS) is a widely used tool for the assessment of the extent and degree of skin thickness. This study aimed to identify the classes of patients with early similar skin thickening trajectories without any a priori assumptions and study their associations with organ involvement and survival. From the French SSc national cohort, patients with a disease duration of less than 2 years at inclusion and with at least 2 mRSS available within the first 4 years of follow-up were enrolled. Classes of patients with similar mRSS trajectories were identified based on a latent class mixed model. The clinical characteristics and survival rate were compared between the obtained classes. A total of 198 patients fulfilled the inclusion criteria, with a total of 641 mRSS available. The median disease duration and follow-up were 0.8 (interquartile range 0.4; 1.2) and 6.3 (3.8; 8.9) years, respectively. Individual trajectories of mRSS were highly heterogeneous between patients. Models with 1-6 latent classes of trajectories were sequentially assessed, and the 5-class model represented the best fit to data. Each class was characterized by a unique global trajectory of mRSS. The median disease duration did not differ significantly between classes. Baseline organ involvement was more frequent in classes with significant change over time (classes 2-5) than in class 1 (low baseline mRSS without significant change over time). Using Cox regression, we observed a progressively increasing risk of death from classes 1 to 5. Early identification of clinical phenotype based on skin thickening trajectories could predict morbi-mortality in SSc. This study suggested that mRSS trajectories characterization might be pivotal for clinical practice and future trial designs.
Sections du résumé
BACKGROUND
Systemic sclerosis (SSc) is a severe and highly heterogeneous disease. The modified Rodnan skin score (mRSS) is a widely used tool for the assessment of the extent and degree of skin thickness. This study aimed to identify the classes of patients with early similar skin thickening trajectories without any a priori assumptions and study their associations with organ involvement and survival.
METHODS
From the French SSc national cohort, patients with a disease duration of less than 2 years at inclusion and with at least 2 mRSS available within the first 4 years of follow-up were enrolled. Classes of patients with similar mRSS trajectories were identified based on a latent class mixed model. The clinical characteristics and survival rate were compared between the obtained classes.
RESULTS
A total of 198 patients fulfilled the inclusion criteria, with a total of 641 mRSS available. The median disease duration and follow-up were 0.8 (interquartile range 0.4; 1.2) and 6.3 (3.8; 8.9) years, respectively. Individual trajectories of mRSS were highly heterogeneous between patients. Models with 1-6 latent classes of trajectories were sequentially assessed, and the 5-class model represented the best fit to data. Each class was characterized by a unique global trajectory of mRSS. The median disease duration did not differ significantly between classes. Baseline organ involvement was more frequent in classes with significant change over time (classes 2-5) than in class 1 (low baseline mRSS without significant change over time). Using Cox regression, we observed a progressively increasing risk of death from classes 1 to 5.
CONCLUSIONS
Early identification of clinical phenotype based on skin thickening trajectories could predict morbi-mortality in SSc. This study suggested that mRSS trajectories characterization might be pivotal for clinical practice and future trial designs.
Identifiants
pubmed: 32070422
doi: 10.1186/s13075-020-2113-6
pii: 10.1186/s13075-020-2113-6
pmc: PMC7029583
doi:
Types de publication
Journal Article
Observational Study
Langues
eng
Sous-ensembles de citation
IM
Pagination
30Investigateurs
Zahir Amoura
(Z)
Olivier Aumaitre
(O)
Eric Auxenfants
(E)
Marie-Hélène Balquet
(MH)
Cristina Belizna
(C)
Boris Bienvenu
(B)
Emmanuel Chatelus
(E)
Robin Dhote
(R)
Yves Frances
(Y)
Jean-Baptiste Gaultier
(JB)
Bernard Imbert
(B)
Jean-Emmanuel Kahn
(JE)
Gilles Kaplanski
(G)
Pierre Kieffer
(P)
Noémie Le Gouellec
(N)
Philippe Guilpain
(P)
Olivier Lidove
(O)
Nadine Magy-Bertrand
(N)
François Maurier
(F)
Thomas Papo
(T)
Jean-Loup Pennaforte
(JL)
Jacques Pouchot
(J)
Vivianne Queyrel
(V)
Denis Wahl
(D)
Références
Rheum Dis Clin North Am. 2003 May;29(2):255-73, vi
pubmed: 12841294
Ann Rheum Dis. 2011 Jan;70(1):104-9
pubmed: 20679474
Ann Rheum Dis. 2010 Oct;69(10):1809-15
pubmed: 20551155
Arthritis Rheumatol. 2019 Nov 4;:
pubmed: 31682743
Arthritis Rheum. 2007 Jul;56(7):2422-31
pubmed: 17599771
Ann Rheum Dis. 2019 May;78(5):648-656
pubmed: 30852552
Arthritis Rheumatol. 2019 Sep;71(9):1553-1570
pubmed: 30969034
Ann Rheum Dis. 2018 Apr;77(4):563-570
pubmed: 29306872
J Rheumatol. 2001 Jul;28(7):1573-6
pubmed: 11469464
Ann Rheum Dis. 2007 Jun;66(6):754-63
pubmed: 17234652
Semin Arthritis Rheum. 2015 Oct;45(2):184-9
pubmed: 25959492
Arthritis Rheumatol. 2020 Jan;72(1):125-136
pubmed: 31342624
Arthritis Rheum. 1980 May;23(5):581-90
pubmed: 7378088
Arthritis Rheum. 2009 Aug;60(8):2490-8
pubmed: 19644851
Rheumatology (Oxford). 2016 Nov;55(11):2001-2008
pubmed: 27520796
Rheumatology (Oxford). 2017 Sep 1;56(suppl_5):v53-v66
pubmed: 28992173
Stat Med. 2002 Feb 15;21(3):417-29
pubmed: 11813228
Autoimmun Rev. 2020 Feb;19(2):102452
pubmed: 31838157
J Rheumatol. 1998 Jan;25(1):84-8
pubmed: 9458208
Int J Rheum Dis. 2019 Jan;22(1):96-102
pubmed: 30398033
Arthritis Rheum. 2013 Nov;65(11):2737-47
pubmed: 24122180
J Scleroderma Relat Disord. 2017 Jan-Apr;2(1):11-18
pubmed: 28516167
Scand J Rheumatol. 2018 Jan;47(1):62-70
pubmed: 28990485
Ann Rheum Dis. 2015 Jun;74(6):1124-31
pubmed: 24981642
Ann Rheum Dis. 2017 Jul;76(7):1207-1218
pubmed: 28188239
Rheumatology (Oxford). 2020 Feb 1;59(2):398-406
pubmed: 31359048
Ann Rheum Dis. 2016 Jan;75(1):103-9
pubmed: 25165035
Ann Rheum Dis. 2012 Aug;71(8):1355-60
pubmed: 22615460
Res Social Adm Pharm. 2017 Nov;13(6):1196-1201
pubmed: 27955976
Ann Rheum Dis. 2007 Jul;66(7):966-9
pubmed: 17234649
Arthritis Res Ther. 2019 Jan 16;21(1):23
pubmed: 30651141
Arthritis Rheum. 1993 Nov;36(11):1575-9
pubmed: 8240434
Arthritis Rheum. 2012 Oct;64(10):3420-9
pubmed: 22328195
Clin Rev Allergy Immunol. 2011 Apr;40(2):78-83
pubmed: 20143182
Arthritis Rheum. 2007 Aug;56(8):2740-6
pubmed: 17665460