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Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)

Identifieur interne : 000129 ( Main/Exploration ); précédent : 000128; suivant : 000130

Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)

Auteurs : Ariane L. Herrick [Royaume-Uni] ; Xiaoyan Pan [Royaume-Uni] ; Sébastien Peytrignet [Royaume-Uni] ; Mark Lunt [Royaume-Uni] ; Roger Hesselstrand [Suède] ; Luc Mouthon [France] ; Alan Silman [Royaume-Uni] ; Edith Brown [Royaume-Uni] ; Lászl Czirják [Hongrie] ; Jörg H W. Distler [Allemagne] ; Oliver Distler [Suisse] ; Kim Fligelstone [Royaume-Uni] ; William J. Gregory [Royaume-Uni] ; Rachel Ochiel [Royaume-Uni] ; Madelon Vonk [Pays-Bas] ; Codrina Ancu A [Roumanie] ; Voon H. Ong [Royaume-Uni] ; Dominique Farge [France] ; Marie Hudson [Canada] ; Marco Matucci-Cerinic [Italie] ; Alexandra Balbir-Gurman [Israël] ; Yvind Midtvedt [Norvège] ; Alison C. Jordan [Royaume-Uni] ; Paresh Jobanputra [Royaume-Uni] ; Wendy Stevens [Australie] ; Pia Moinzadeh [Allemagne] ; Frances C. Hall [Royaume-Uni] ; Christian Agard [France] ; Marina E. Anderson [Royaume-Uni] ; Elisabeth Diot [France] ; Rajan Madhok [Royaume-Uni] ; Mohammed Akil [Royaume-Uni] ; Maya H. Buch [Royaume-Uni] ; Lorinda Chung [États-Unis] ; Nemanja Damjanov [Serbie] ; Harsha Gunawardena [Royaume-Uni] ; Peter Lanyon [Royaume-Uni] ; Yasmeen Ahmad [Royaume-Uni] ; Kuntal Chakravarty [Royaume-Uni] ; S Ren Jacobsen [Danemark] ; Alexander J. Macgregor [Royaume-Uni] ; Neil Mchugh [Royaume-Uni] ; Ulf Müller-Ladner [Allemagne] ; Gabriela Riemekasten [Allemagne] ; Michael Becker [Allemagne] ; Janet Roddy [Australie] ; Patricia E. Carreira [Espagne] ; Anne Laure Fauchais [France] ; Eric Hachulla [France] ; Jennifer Hamilton [Royaume-Uni] ; Murat Nanç [Turquie] ; John S. Mclaren [Royaume-Uni] ; Jacob M. Van Laar [Pays-Bas] ; Sanjay Pathare [Royaume-Uni] ; Susannah Proudman ; Anna Rudin [Suède] ; Joanne Sahhar [Australie] ; Brigitte Coppere [France] ; Christine Serratrice [France] ; Tom Sheeran [Royaume-Uni] ; Douglas J. Veale [Irlande (pays)] ; Claire Grange [France] ; Georges-Selim Trad [France] ; Christopher P. Denton [Royaume-Uni]

Source :

RBID : PMC:5530354

Descripteurs français

English descriptors

Abstract

Objectives

The rarity of early diffuse cutaneous systemic sclerosis (dcSSc) makes randomised controlled trials very difficult. We aimed to use an observational approach to compare effectiveness of currently used treatment approaches.

Methods

This was a prospective, observational cohort study of early dcSSc (within three years of onset of skin thickening). Clinicians selected one of four protocols for each patient: methotrexate, mycophenolate mofetil (MMF), cyclophosphamide or ‘no immunosuppressant’. Patients were assessed three-monthly for up to 24 months. The primary outcome was the change in modified Rodnan skin score (mRSS). Confounding by indication at baseline was accounted for using inverse probability of treatment (IPT) weights. As a secondary outcome, an IPT-weighted Cox model was used to test for differences in survival.

Results

Of 326 patients recruited from 50 centres, 65 were prescribed methotrexate, 118 MMF, 87 cyclophosphamide and 56 no immunosuppressant. 276 (84.7%) patients completed 12 and 234 (71.7%) 24 months follow-up (or reached last visit date). There were statistically significant reductions in mRSS at 12 months in all groups: −4.0 (−5.2 to −2.7) units for methotrexate, −4.1 (−5.3 to −2.9) for MMF, −3.3 (−4.9 to −1.7) for cyclophosphamide and −2.2 (−4.0 to −0.3) for no immunosuppressant (p value for between-group differences=0.346). There were no statistically significant differences in survival between protocols before (p=0.389) or after weighting (p=0.440), but survival was poorest in the no immunosuppressant group (84.0%) at 24 months.

Conclusions

These findings may support using immunosuppressants for early dcSSc but suggest that overall benefit is modest over 12 months and that better treatments are needed.

Trial registration number

NCT02339441.


Url:
DOI: 10.1136/annrheumdis-2016-210503
PubMed: 28188239
PubMed Central: 5530354


Affiliations:


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<name sortKey="Hesselstrand, Roger" sort="Hesselstrand, Roger" uniqKey="Hesselstrand R" first="Roger" last="Hesselstrand">Roger Hesselstrand</name>
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<addr-line>Lund</addr-line>
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<country xml:lang="fr">France</country>
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<name sortKey="Silman, Alan" sort="Silman, Alan" uniqKey="Silman A" first="Alan" last="Silman">Alan Silman</name>
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<addr-line>Oxford</addr-line>
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<country>UK</country>
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<country xml:lang="fr">Royaume-Uni</country>
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<addr-line>Manchester</addr-line>
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<country xml:lang="fr">Royaume-Uni</country>
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<name sortKey="Czirjak, Laszl" sort="Czirjak, Laszl" uniqKey="Czirjak L" first="Lászl" last="Czirják">Lászl Czirják</name>
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<addr-line>Pecs</addr-line>
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<country>Hungary</country>
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<country xml:lang="fr">Hongrie</country>
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<name sortKey="Distler, Jorg H W" sort="Distler, Jorg H W" uniqKey="Distler J" first="Jörg H W" last="Distler">Jörg H W. Distler</name>
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<nlm:aff id="af9">
<addr-line>Department of Internal Medicine 3</addr-line>
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<institution>University of Erlangen-Nuremberg</institution>
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<addr-line>Erlangen</addr-line>
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<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<name sortKey="Distler, Oliver" sort="Distler, Oliver" uniqKey="Distler O" first="Oliver" last="Distler">Oliver Distler</name>
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<addr-line>Department of Rheumatology</addr-line>
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<institution>University of Zurich</institution>
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<addr-line>Zurich</addr-line>
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<country xml:lang="fr">Suisse</country>
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<name sortKey="Fligelstone, Kim" sort="Fligelstone, Kim" uniqKey="Fligelstone K" first="Kim" last="Fligelstone">Kim Fligelstone</name>
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<addr-line>London</addr-line>
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<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<name sortKey="Gregory, William J" sort="Gregory, William J" uniqKey="Gregory W" first="William J" last="Gregory">William J. Gregory</name>
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<addr-line>Rehabilitation Services</addr-line>
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<institution>Salford Royal NHS Foundation Trust</institution>
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<addr-line>Salford</addr-line>
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<country xml:lang="fr">Royaume-Uni</country>
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<name sortKey="Ochiel, Rachel" sort="Ochiel, Rachel" uniqKey="Ochiel R" first="Rachel" last="Ochiel">Rachel Ochiel</name>
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<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<name sortKey="Vonk, Madelon" sort="Vonk, Madelon" uniqKey="Vonk M" first="Madelon" last="Vonk">Madelon Vonk</name>
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<addr-line>Department of the Rheumatic Diseases</addr-line>
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<addr-line>Nijmegen</addr-line>
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<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<author>
<name sortKey="Ancu A, Codrina" sort="Ancu A, Codrina" uniqKey="Ancu A C" first="Codrina" last="Ancu A">Codrina Ancu A</name>
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<addr-line>Rheumatology 2 Department</addr-line>
,
<institution>“Grigore T. Popa” University of Medicine and Pharmacy, Clinical Rehabilitation Hospital</institution>
,
<addr-line>Iași</addr-line>
,
<country>Romania</country>
</nlm:aff>
<country xml:lang="fr">Roumanie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<name sortKey="Ong, Voon H" sort="Ong, Voon H" uniqKey="Ong V" first="Voon H" last="Ong">Voon H. Ong</name>
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<name sortKey="Farge, Dominique" sort="Farge, Dominique" uniqKey="Farge D" first="Dominique" last="Farge">Dominique Farge</name>
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<addr-line>Unité Clinique de Médecine Interne</addr-line>
,
<institution>Maladies Auto-immunes et Pathologie Vasculaire, UF 04, Hôpital Saint-Louis, AP-HP Assistance Publique des Hôpitaux de Paris, INSERM UMRS 1160, Paris Denis Diderot University,</institution>
<country>France</country>
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<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<author>
<name sortKey="Hudson, Marie" sort="Hudson, Marie" uniqKey="Hudson M" first="Marie" last="Hudson">Marie Hudson</name>
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<nlm:aff id="af17">
<institution>Jewish General Hospital, Lady Davis Institute and McGill University</institution>
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<addr-line>Montreal</addr-line>
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<country>Canada</country>
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<country xml:lang="fr">Canada</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<author>
<name sortKey="Matucci Cerinic, Marco" sort="Matucci Cerinic, Marco" uniqKey="Matucci Cerinic M" first="Marco" last="Matucci-Cerinic">Marco Matucci-Cerinic</name>
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<nlm:aff id="af18">
<addr-line>Department Experimental and Clinical Medicine, Division of Rheumatology AOUC</addr-line>
,
<institution>University of Florence</institution>
,
<addr-line>Florence</addr-line>
,
<country>Italy</country>
</nlm:aff>
<country xml:lang="fr">Italie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Balbir Gurman, Alexandra" sort="Balbir Gurman, Alexandra" uniqKey="Balbir Gurman A" first="Alexandra" last="Balbir-Gurman">Alexandra Balbir-Gurman</name>
<affiliation wicri:level="1">
<nlm:aff id="af19">
<addr-line>Shine Rheumatology Unit</addr-line>
,
<institution>Rambam Heath Care Campus; Rappaport Faculty of Medicine</institution>
,
<addr-line>Haifa</addr-line>
,
<country>Israel</country>
</nlm:aff>
<country xml:lang="fr">Israël</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Midtvedt, Yvind" sort="Midtvedt, Yvind" uniqKey="Midtvedt " first=" Yvind" last="Midtvedt"> Yvind Midtvedt</name>
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<nlm:aff id="af20">
<addr-line>Rheumatology Unit</addr-line>
,
<institution>Oslo University Hospital Rikshospitalet</institution>
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<addr-line>Oslo</addr-line>
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<country>Norway</country>
</nlm:aff>
<country xml:lang="fr">Norvège</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jordan, Alison C" sort="Jordan, Alison C" uniqKey="Jordan A" first="Alison C" last="Jordan">Alison C. Jordan</name>
<affiliation wicri:level="1">
<nlm:aff id="af21">
<institution>Queen Elizabeth Hospital Birmingham, UHB Foundation Trust</institution>
,
<addr-line>Birmingham</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jobanputra, Paresh" sort="Jobanputra, Paresh" uniqKey="Jobanputra P" first="Paresh" last="Jobanputra">Paresh Jobanputra</name>
<affiliation wicri:level="1">
<nlm:aff id="af21">
<institution>Queen Elizabeth Hospital Birmingham, UHB Foundation Trust</institution>
,
<addr-line>Birmingham</addr-line>
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<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Stevens, Wendy" sort="Stevens, Wendy" uniqKey="Stevens W" first="Wendy" last="Stevens">Wendy Stevens</name>
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<nlm:aff id="af22">
<institution>St Vincent's Hospital</institution>
,
<addr-line>Melbourne</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Moinzadeh, Pia" sort="Moinzadeh, Pia" uniqKey="Moinzadeh P" first="Pia" last="Moinzadeh">Pia Moinzadeh</name>
<affiliation wicri:level="1">
<nlm:aff id="af23">
<addr-line>Department for Dermatology</addr-line>
,
<institution>University of Cologne Kerpenerstr. 62</institution>
,
<addr-line>Köln</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Hall, Frances C" sort="Hall, Frances C" uniqKey="Hall F" first="Frances C" last="Hall">Frances C. Hall</name>
<affiliation wicri:level="1">
<nlm:aff id="af24">
<institution>Cambridge University NHS Hospital Foundation Trust</institution>
,
<addr-line>Cambridge</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Agard, Christian" sort="Agard, Christian" uniqKey="Agard C" first="Christian" last="Agard">Christian Agard</name>
<affiliation wicri:level="1">
<nlm:aff id="af25">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Hôtel-Dieu Hospital</institution>
,
<institution>University of Nantes</institution>
,
<addr-line>Nantes</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
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<author>
<name sortKey="Anderson, Marina E" sort="Anderson, Marina E" uniqKey="Anderson M" first="Marina E" last="Anderson">Marina E. Anderson</name>
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<nlm:aff id="af26">
<institution>University of Liverpool, Aintree University Hospital</institution>
,
<addr-line>Liverpool</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Diot, Elisabeth" sort="Diot, Elisabeth" uniqKey="Diot E" first="Elisabeth" last="Diot">Elisabeth Diot</name>
<affiliation wicri:level="1">
<nlm:aff id="af27">
<addr-line>Service de Médecine Interne</addr-line>
,
<institution>Hôpital Bretonneau Tours</institution>
<addr-line>Cedex</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Madhok, Rajan" sort="Madhok, Rajan" uniqKey="Madhok R" first="Rajan" last="Madhok">Rajan Madhok</name>
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<nlm:aff id="af28">
<institution>Centre for Rheumatic Diseases</institution>
,
<addr-line>Glasgow Royal Infirmary, Glasgow</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
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<author>
<name sortKey="Akil, Mohammed" sort="Akil, Mohammed" uniqKey="Akil M" first="Mohammed" last="Akil">Mohammed Akil</name>
<affiliation wicri:level="1">
<nlm:aff id="af29">
<institution>Sheffield Teaching Hospitals</institution>
,
<addr-line>Sheffield</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Buch, Maya H" sort="Buch, Maya H" uniqKey="Buch M" first="Maya H" last="Buch">Maya H. Buch</name>
<affiliation wicri:level="1">
<nlm:aff id="af30">
<institution>Leeds Institute of Rheumatic and Musculoskeletal Medicine, University of Leeds and NIHR Leeds Musculoskeletal Biomedical Research Unit, Leeds Teaching Hospitals NHS Trust</institution>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Chung, Lorinda" sort="Chung, Lorinda" uniqKey="Chung L" first="Lorinda" last="Chung">Lorinda Chung</name>
<affiliation wicri:level="1">
<nlm:aff id="af31">
<institution>Stanford University</institution>
,
<addr-line>Stanford, California</addr-line>
,
<country>USA</country>
.</nlm:aff>
<country xml:lang="fr">États-Unis</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Damjanov, Nemanja" sort="Damjanov, Nemanja" uniqKey="Damjanov N" first="Nemanja" last="Damjanov">Nemanja Damjanov</name>
<affiliation wicri:level="1">
<nlm:aff id="af32">
<institution>University of Belgrade School of Medicine, Institute of Rheumatology</institution>
,
<addr-line>Belgrade</addr-line>
,
<country>Serbia</country>
</nlm:aff>
<country xml:lang="fr">Serbie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Gunawardena, Harsha" sort="Gunawardena, Harsha" uniqKey="Gunawardena H" first="Harsha" last="Gunawardena">Harsha Gunawardena</name>
<affiliation wicri:level="1">
<nlm:aff id="af33">
<institution>Clinical and Academic Rheumatology, North Bristol NHS Trust</institution>
,
<addr-line>Bristol</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Lanyon, Peter" sort="Lanyon, Peter" uniqKey="Lanyon P" first="Peter" last="Lanyon">Peter Lanyon</name>
<affiliation wicri:level="1">
<nlm:aff id="af34">
<institution>Nottingham University Hospitals NHS Trust, and Nottingham NHS Treatment Centre</institution>
,
<addr-line>Nottingham</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Ahmad, Yasmeen" sort="Ahmad, Yasmeen" uniqKey="Ahmad Y" first="Yasmeen" last="Ahmad">Yasmeen Ahmad</name>
<affiliation wicri:level="1">
<nlm:aff id="af35">
<institution>Peter Maddison Rheumatology Centre</institution>
,
<addr-line>Llandudno</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Chakravarty, Kuntal" sort="Chakravarty, Kuntal" uniqKey="Chakravarty K" first="Kuntal" last="Chakravarty">Kuntal Chakravarty</name>
<affiliation wicri:level="1">
<nlm:aff id="af36">
<institution>Queens Hospital</institution>
,
<addr-line>Romford</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jacobsen, S Ren" sort="Jacobsen, S Ren" uniqKey="Jacobsen S" first="S Ren" last="Jacobsen">S Ren Jacobsen</name>
<affiliation wicri:level="1">
<nlm:aff id="af37">
<institution>University of Copenhagen, Copenhagen Lupus and Vasculitis Clinic, Center for Rheumatology and Spine Diseases, Rigshospitalet</institution>
,
<addr-line>Copenhagen</addr-line>
,
<country>Denmark</country>
</nlm:aff>
<country xml:lang="fr">Danemark</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Macgregor, Alexander J" sort="Macgregor, Alexander J" uniqKey="Macgregor A" first="Alexander J" last="Macgregor">Alexander J. Macgregor</name>
<affiliation wicri:level="1">
<nlm:aff id="af38">
<institution>Norwich Medical School, University of East Anglia</institution>
,
<addr-line>Norwich</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Mchugh, Neil" sort="Mchugh, Neil" uniqKey="Mchugh N" first="Neil" last="Mchugh">Neil Mchugh</name>
<affiliation wicri:level="1">
<nlm:aff id="af39">
<institution>Royal National Hospital for Rheumatic Diseases</institution>
,
<addr-line>Bath</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Muller Ladner, Ulf" sort="Muller Ladner, Ulf" uniqKey="Muller Ladner U" first="Ulf" last="Müller-Ladner">Ulf Müller-Ladner</name>
<affiliation wicri:level="1">
<nlm:aff id="af40">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>Justus-Liebig University Giessen</institution>
,
<addr-line>Bad Nauheim</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Riemekasten, Gabriela" sort="Riemekasten, Gabriela" uniqKey="Riemekasten G" first="Gabriela" last="Riemekasten">Gabriela Riemekasten</name>
<affiliation wicri:level="1">
<nlm:aff id="af41">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>University of Lübeck</institution>
,
<addr-line>Lübeck</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Becker, Michael" sort="Becker, Michael" uniqKey="Becker M" first="Michael" last="Becker">Michael Becker</name>
<affiliation wicri:level="1">
<nlm:aff id="af42">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>University Hospital Charité Berlin</institution>
,
<addr-line>Berlin</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Roddy, Janet" sort="Roddy, Janet" uniqKey="Roddy J" first="Janet" last="Roddy">Janet Roddy</name>
<affiliation wicri:level="1">
<nlm:aff id="af43">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>Royal Perth Hospital</institution>
,
<addr-line>Perth</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Carreira, Patricia E" sort="Carreira, Patricia E" uniqKey="Carreira P" first="Patricia E" last="Carreira">Patricia E. Carreira</name>
<affiliation wicri:level="1">
<nlm:aff id="af44">
<institution>Servicio de Reumatologia. Hospital Universitario 12 de Octubre</institution>
,
<addr-line>Madrid</addr-line>
,
<country>Spain</country>
</nlm:aff>
<country xml:lang="fr">Espagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Fauchais, Anne Laure" sort="Fauchais, Anne Laure" uniqKey="Fauchais A" first="Anne Laure" last="Fauchais">Anne Laure Fauchais</name>
<affiliation wicri:level="1">
<nlm:aff id="af45">
<addr-line>Internal Medicine Unit</addr-line>
,
<institution>Limoges University Hospital</institution>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hachulla, Eric" sort="Hachulla, Eric" uniqKey="Hachulla E" first="Eric" last="Hachulla">Eric Hachulla</name>
<affiliation wicri:level="1">
<nlm:aff id="af46">
<addr-line>Département de Médecine Interne et Immunologie Clinique</addr-line>
,
<institution>Centre National de Référence Maladies Systémiques etAuto-immunes Rares, Université de Lille, Inserm, U995, FHU Immune-Mediated Inflammatory Diseases and Targeted Therapies,</institution>
<addr-line>Lille</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hamilton, Jennifer" sort="Hamilton, Jennifer" uniqKey="Hamilton J" first="Jennifer" last="Hamilton">Jennifer Hamilton</name>
<affiliation wicri:level="1">
<nlm:aff id="af47">
<institution>Gateshead Hospitals Foundation Trust</institution>
,
<addr-line>Gateshead</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey=" Nanc, Murat" sort=" Nanc, Murat" uniqKey=" Nanc M" first="Murat" last=" Nanç">Murat Nanç</name>
<affiliation wicri:level="1">
<nlm:aff id="af48">
<addr-line>Department of Internal Medicine, Division of Rheumatology</addr-line>
,
<institution>Istanbul University</institution>
,
<addr-line>Istanbul</addr-line>
,
<country>Turkey</country>
</nlm:aff>
<country xml:lang="fr">Turquie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Mclaren, John S" sort="Mclaren, John S" uniqKey="Mclaren J" first="John S" last="Mclaren">John S. Mclaren</name>
<affiliation wicri:level="1">
<nlm:aff id="af49">
<addr-line>Fife Rheumatic Diseases Unit</addr-line>
,
<institution>Whyteman's Brae Hospital</institution>
,
<addr-line>Kirkcaldy</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Van Laar, Jacob M" sort="Van Laar, Jacob M" uniqKey="Van Laar J" first="Jacob M" last="Van Laar">Jacob M. Van Laar</name>
<affiliation wicri:level="1">
<nlm:aff id="af50">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>UMC Utrecht</institution>
,
<addr-line>Utrecht</addr-line>
,
<country>The Netherlands</country>
</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Pathare, Sanjay" sort="Pathare, Sanjay" uniqKey="Pathare S" first="Sanjay" last="Pathare">Sanjay Pathare</name>
<affiliation wicri:level="1">
<nlm:aff id="af51">
<institution>James Cook University Hospital</institution>
,
<addr-line>Middlesbrough</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Proudman, Susannah" sort="Proudman, Susannah" uniqKey="Proudman S" first="Susannah" last="Proudman">Susannah Proudman</name>
<affiliation>
<nlm:aff id="af52">
<addr-line>Rheumatology Unit</addr-line>
,
<institution>Royal Adelaide Hospital, and Discipline of Medicine, University of Adelaide</institution>
,
<addr-line>Adelaide</addr-line>
,
<country>South Australia</country>
</nlm:aff>
<wicri:noCountry code="nlm country">South Australia</wicri:noCountry>
</affiliation>
</author>
<author>
<name sortKey="Rudin, Anna" sort="Rudin, Anna" uniqKey="Rudin A" first="Anna" last="Rudin">Anna Rudin</name>
<affiliation wicri:level="1">
<nlm:aff id="af53">
<addr-line>Department of Rheumatology and Inflammation Research</addr-line>
,
<institution>The Sahlgrenska Academy at Gothenburg University</institution>
,
<addr-line>Gothenburg</addr-line>
,
<country>Sweden</country>
</nlm:aff>
<country xml:lang="fr">Suède</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Sahhar, Joanne" sort="Sahhar, Joanne" uniqKey="Sahhar J" first="Joanne" last="Sahhar">Joanne Sahhar</name>
<affiliation wicri:level="1">
<nlm:aff id="af54">
<institution>Monash Centre for Inflammatory Diseases, Monash University</institution>
,
<addr-line>Clayton, Melbourne</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Coppere, Brigitte" sort="Coppere, Brigitte" uniqKey="Coppere B" first="Brigitte" last="Coppere">Brigitte Coppere</name>
<affiliation wicri:level="1">
<nlm:aff id="af55">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Hôpital Edouard Herriot</institution>
,
<addr-line>Lyon</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Serratrice, Christine" sort="Serratrice, Christine" uniqKey="Serratrice C" first="Christine" last="Serratrice">Christine Serratrice</name>
<affiliation wicri:level="1">
<nlm:aff id="af56">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Foundation Hospital Saint Joseph</institution>
,
<addr-line>Marseille</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Sheeran, Tom" sort="Sheeran, Tom" uniqKey="Sheeran T" first="Tom" last="Sheeran">Tom Sheeran</name>
<affiliation wicri:level="1">
<nlm:aff id="af57">
<institution>Cannock Chase Hospital</institution>
,
<addr-line>Cannock</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Veale, Douglas J" sort="Veale, Douglas J" uniqKey="Veale D" first="Douglas J" last="Veale">Douglas J. Veale</name>
<affiliation wicri:level="1">
<nlm:aff id="af58">
<institution>St Vincent's University Hospital</institution>
,
<addr-line>Dublin</addr-line>
,
<country>Ireland</country>
</nlm:aff>
<country xml:lang="fr">Irlande (pays)</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Grange, Claire" sort="Grange, Claire" uniqKey="Grange C" first="Claire" last="Grange">Claire Grange</name>
<affiliation wicri:level="1">
<nlm:aff id="af59">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Centre Hospitalier Lyon Sud</institution>
,
<addr-line>Pierre Benite</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Trad, Georges Selim" sort="Trad, Georges Selim" uniqKey="Trad G" first="Georges-Selim" last="Trad">Georges-Selim Trad</name>
<affiliation wicri:level="1">
<nlm:aff id="af60">
<addr-line>Internal Medecine</addr-line>
,
<institution>Ambroise Paré Hospital</institution>
,
<addr-line>Boulogne Billancourt</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Denton, Christopher P" sort="Denton, Christopher P" uniqKey="Denton C" first="Christopher P" last="Denton">Christopher P. Denton</name>
<affiliation wicri:level="1">
<nlm:aff id="af15">
<addr-line>UCL Division of Medicine</addr-line>
,
<institution>Centre for Rheumatology and Connective Tissue Diseases</institution>
,
<addr-line>London</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
</titleStmt>
<publicationStmt>
<idno type="wicri:source">PMC</idno>
<idno type="pmid">28188239</idno>
<idno type="pmc">5530354</idno>
<idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5530354</idno>
<idno type="RBID">PMC:5530354</idno>
<idno type="doi">10.1136/annrheumdis-2016-210503</idno>
<date when="2017">2017</date>
<idno type="wicri:Area/Pmc/Corpus">002260</idno>
<idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002260</idno>
<idno type="wicri:Area/Pmc/Curation">002110</idno>
<idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">002110</idno>
<idno type="wicri:Area/Pmc/Checkpoint">000026</idno>
<idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">000026</idno>
<idno type="wicri:source">PubMed</idno>
<idno type="wicri:Area/PubMed/Corpus">000A03</idno>
<idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">000A03</idno>
<idno type="wicri:Area/PubMed/Curation">000A00</idno>
<idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Curation">000A00</idno>
<idno type="wicri:Area/PubMed/Checkpoint">000A00</idno>
<idno type="wicri:explorRef" wicri:stream="Checkpoint" wicri:step="PubMed">000A00</idno>
<idno type="wicri:Area/Ncbi/Merge">004302</idno>
<idno type="wicri:Area/Ncbi/Curation">004302</idno>
<idno type="wicri:Area/Ncbi/Checkpoint">004302</idno>
<idno type="wicri:doubleKey">0003-4967:2017:Herrick A:treatment:outcome:in</idno>
<idno type="wicri:Area/Main/Merge">000124</idno>
<idno type="wicri:Area/Main/Curation">000129</idno>
<idno type="wicri:Area/Main/Exploration">000129</idno>
</publicationStmt>
<sourceDesc>
<biblStruct>
<analytic>
<title xml:lang="en" level="a" type="main">Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)</title>
<author>
<name sortKey="Herrick, Ariane L" sort="Herrick, Ariane L" uniqKey="Herrick A" first="Ariane L" last="Herrick">Ariane L. Herrick</name>
<affiliation wicri:level="1">
<nlm:aff id="af1">
<institution>Centre for Musculoskeletal Research, The University of Manchester, Salford Royal NHS Foundation Trust, Manchester Academic Health Science Centre</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
<affiliation wicri:level="1">
<nlm:aff id="af2">
<addr-line>NIHR Manchester Musculoskeletal Biomedical Research Unit</addr-line>
,
<institution>Central Manchester NHS Foundation Trust, Manchester Academic Health Science Centre</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Pan, Xiaoyan" sort="Pan, Xiaoyan" uniqKey="Pan X" first="Xiaoyan" last="Pan">Xiaoyan Pan</name>
<affiliation wicri:level="1">
<nlm:aff id="af3">
<institution>Centre for Musculoskeletal Research, The University of Manchester, Manchester Academic Health Science Centre</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Peytrignet, Sebastien" sort="Peytrignet, Sebastien" uniqKey="Peytrignet S" first="Sébastien" last="Peytrignet">Sébastien Peytrignet</name>
<affiliation wicri:level="1">
<nlm:aff id="af3">
<institution>Centre for Musculoskeletal Research, The University of Manchester, Manchester Academic Health Science Centre</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Lunt, Mark" sort="Lunt, Mark" uniqKey="Lunt M" first="Mark" last="Lunt">Mark Lunt</name>
<affiliation wicri:level="1">
<nlm:aff id="af3">
<institution>Centre for Musculoskeletal Research, The University of Manchester, Manchester Academic Health Science Centre</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hesselstrand, Roger" sort="Hesselstrand, Roger" uniqKey="Hesselstrand R" first="Roger" last="Hesselstrand">Roger Hesselstrand</name>
<affiliation wicri:level="1">
<nlm:aff id="af4">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>Lund University</institution>
,
<addr-line>Lund</addr-line>
,
<country>Sweden</country>
</nlm:aff>
<country xml:lang="fr">Suède</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Mouthon, Luc" sort="Mouthon, Luc" uniqKey="Mouthon L" first="Luc" last="Mouthon">Luc Mouthon</name>
<affiliation wicri:level="1">
<nlm:aff id="af5">
<addr-line>Service de Médecine Interne</addr-line>
,
<institution>Hôpital Cochin, Centre de Référence pour les Vascularites Nécrosantes et la Sclérodermie Systémique, Université Paris Descartes, Assistance Publique-Hôpitaux de Paris (AP-HP)</institution>
,
<addr-line>Paris</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Silman, Alan" sort="Silman, Alan" uniqKey="Silman A" first="Alan" last="Silman">Alan Silman</name>
<affiliation wicri:level="1">
<nlm:aff id="af6">
<addr-line>Nuffield Department of Orthopaedics</addr-line>
,
<institution>Rheumatology and Musculoskeletal Sciences,University of Oxford</institution>
,
<addr-line>Oxford</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Brown, Edith" sort="Brown, Edith" uniqKey="Brown E" first="Edith" last="Brown">Edith Brown</name>
<affiliation wicri:level="1">
<nlm:aff id="af7">
<institution>Member of Steering Committee, contact via Professor Herrick, The University of Manchester</institution>
,
<addr-line>Manchester</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Czirjak, Laszl" sort="Czirjak, Laszl" uniqKey="Czirjak L" first="Lászl" last="Czirják">Lászl Czirják</name>
<affiliation wicri:level="1">
<nlm:aff id="af8">
<addr-line>Department of Rheumatology and Immunology</addr-line>
,
<institution>Medical Center, University of Pécs</institution>
,
<addr-line>Pecs</addr-line>
,
<country>Hungary</country>
</nlm:aff>
<country xml:lang="fr">Hongrie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Distler, Jorg H W" sort="Distler, Jorg H W" uniqKey="Distler J" first="Jörg H W" last="Distler">Jörg H W. Distler</name>
<affiliation wicri:level="1">
<nlm:aff id="af9">
<addr-line>Department of Internal Medicine 3</addr-line>
,
<institution>University of Erlangen-Nuremberg</institution>
,
<addr-line>Erlangen</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Distler, Oliver" sort="Distler, Oliver" uniqKey="Distler O" first="Oliver" last="Distler">Oliver Distler</name>
<affiliation wicri:level="1">
<nlm:aff id="af10">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>University of Zurich</institution>
,
<addr-line>Zurich</addr-line>
,
<country>Switzerland</country>
</nlm:aff>
<country xml:lang="fr">Suisse</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Fligelstone, Kim" sort="Fligelstone, Kim" uniqKey="Fligelstone K" first="Kim" last="Fligelstone">Kim Fligelstone</name>
<affiliation wicri:level="1">
<nlm:aff id="af11">
<institution>Royal Free London NHS Foundation Trust</institution>
,
<addr-line>London</addr-line>
,
<country>UK</country>
.</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Gregory, William J" sort="Gregory, William J" uniqKey="Gregory W" first="William J" last="Gregory">William J. Gregory</name>
<affiliation wicri:level="1">
<nlm:aff id="af12">
<addr-line>Rehabilitation Services</addr-line>
,
<institution>Salford Royal NHS Foundation Trust</institution>
,
<addr-line>Salford</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Ochiel, Rachel" sort="Ochiel, Rachel" uniqKey="Ochiel R" first="Rachel" last="Ochiel">Rachel Ochiel</name>
<affiliation wicri:level="1">
<nlm:aff id="af11">
<institution>Royal Free London NHS Foundation Trust</institution>
,
<addr-line>London</addr-line>
,
<country>UK</country>
.</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Vonk, Madelon" sort="Vonk, Madelon" uniqKey="Vonk M" first="Madelon" last="Vonk">Madelon Vonk</name>
<affiliation wicri:level="1">
<nlm:aff id="af13">
<addr-line>Department of the Rheumatic Diseases</addr-line>
,
<institution>Radboud University Nijmegen Medical Centre</institution>
,
<addr-line>Nijmegen</addr-line>
,
<country>The Netherlands</country>
</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Ancu A, Codrina" sort="Ancu A, Codrina" uniqKey="Ancu A C" first="Codrina" last="Ancu A">Codrina Ancu A</name>
<affiliation wicri:level="1">
<nlm:aff id="af14">
<addr-line>Rheumatology 2 Department</addr-line>
,
<institution>“Grigore T. Popa” University of Medicine and Pharmacy, Clinical Rehabilitation Hospital</institution>
,
<addr-line>Iași</addr-line>
,
<country>Romania</country>
</nlm:aff>
<country xml:lang="fr">Roumanie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Ong, Voon H" sort="Ong, Voon H" uniqKey="Ong V" first="Voon H" last="Ong">Voon H. Ong</name>
<affiliation wicri:level="1">
<nlm:aff id="af15">
<addr-line>UCL Division of Medicine</addr-line>
,
<institution>Centre for Rheumatology and Connective Tissue Diseases</institution>
,
<addr-line>London</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Farge, Dominique" sort="Farge, Dominique" uniqKey="Farge D" first="Dominique" last="Farge">Dominique Farge</name>
<affiliation wicri:level="1">
<nlm:aff id="af16">
<addr-line>Unité Clinique de Médecine Interne</addr-line>
,
<institution>Maladies Auto-immunes et Pathologie Vasculaire, UF 04, Hôpital Saint-Louis, AP-HP Assistance Publique des Hôpitaux de Paris, INSERM UMRS 1160, Paris Denis Diderot University,</institution>
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hudson, Marie" sort="Hudson, Marie" uniqKey="Hudson M" first="Marie" last="Hudson">Marie Hudson</name>
<affiliation wicri:level="1">
<nlm:aff id="af17">
<institution>Jewish General Hospital, Lady Davis Institute and McGill University</institution>
,
<addr-line>Montreal</addr-line>
,
<country>Canada</country>
</nlm:aff>
<country xml:lang="fr">Canada</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Matucci Cerinic, Marco" sort="Matucci Cerinic, Marco" uniqKey="Matucci Cerinic M" first="Marco" last="Matucci-Cerinic">Marco Matucci-Cerinic</name>
<affiliation wicri:level="1">
<nlm:aff id="af18">
<addr-line>Department Experimental and Clinical Medicine, Division of Rheumatology AOUC</addr-line>
,
<institution>University of Florence</institution>
,
<addr-line>Florence</addr-line>
,
<country>Italy</country>
</nlm:aff>
<country xml:lang="fr">Italie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Balbir Gurman, Alexandra" sort="Balbir Gurman, Alexandra" uniqKey="Balbir Gurman A" first="Alexandra" last="Balbir-Gurman">Alexandra Balbir-Gurman</name>
<affiliation wicri:level="1">
<nlm:aff id="af19">
<addr-line>Shine Rheumatology Unit</addr-line>
,
<institution>Rambam Heath Care Campus; Rappaport Faculty of Medicine</institution>
,
<addr-line>Haifa</addr-line>
,
<country>Israel</country>
</nlm:aff>
<country xml:lang="fr">Israël</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Midtvedt, Yvind" sort="Midtvedt, Yvind" uniqKey="Midtvedt " first=" Yvind" last="Midtvedt"> Yvind Midtvedt</name>
<affiliation wicri:level="1">
<nlm:aff id="af20">
<addr-line>Rheumatology Unit</addr-line>
,
<institution>Oslo University Hospital Rikshospitalet</institution>
,
<addr-line>Oslo</addr-line>
,
<country>Norway</country>
</nlm:aff>
<country xml:lang="fr">Norvège</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jordan, Alison C" sort="Jordan, Alison C" uniqKey="Jordan A" first="Alison C" last="Jordan">Alison C. Jordan</name>
<affiliation wicri:level="1">
<nlm:aff id="af21">
<institution>Queen Elizabeth Hospital Birmingham, UHB Foundation Trust</institution>
,
<addr-line>Birmingham</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jobanputra, Paresh" sort="Jobanputra, Paresh" uniqKey="Jobanputra P" first="Paresh" last="Jobanputra">Paresh Jobanputra</name>
<affiliation wicri:level="1">
<nlm:aff id="af21">
<institution>Queen Elizabeth Hospital Birmingham, UHB Foundation Trust</institution>
,
<addr-line>Birmingham</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Stevens, Wendy" sort="Stevens, Wendy" uniqKey="Stevens W" first="Wendy" last="Stevens">Wendy Stevens</name>
<affiliation wicri:level="1">
<nlm:aff id="af22">
<institution>St Vincent's Hospital</institution>
,
<addr-line>Melbourne</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Moinzadeh, Pia" sort="Moinzadeh, Pia" uniqKey="Moinzadeh P" first="Pia" last="Moinzadeh">Pia Moinzadeh</name>
<affiliation wicri:level="1">
<nlm:aff id="af23">
<addr-line>Department for Dermatology</addr-line>
,
<institution>University of Cologne Kerpenerstr. 62</institution>
,
<addr-line>Köln</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hall, Frances C" sort="Hall, Frances C" uniqKey="Hall F" first="Frances C" last="Hall">Frances C. Hall</name>
<affiliation wicri:level="1">
<nlm:aff id="af24">
<institution>Cambridge University NHS Hospital Foundation Trust</institution>
,
<addr-line>Cambridge</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Agard, Christian" sort="Agard, Christian" uniqKey="Agard C" first="Christian" last="Agard">Christian Agard</name>
<affiliation wicri:level="1">
<nlm:aff id="af25">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Hôtel-Dieu Hospital</institution>
,
<institution>University of Nantes</institution>
,
<addr-line>Nantes</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Anderson, Marina E" sort="Anderson, Marina E" uniqKey="Anderson M" first="Marina E" last="Anderson">Marina E. Anderson</name>
<affiliation wicri:level="1">
<nlm:aff id="af26">
<institution>University of Liverpool, Aintree University Hospital</institution>
,
<addr-line>Liverpool</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Diot, Elisabeth" sort="Diot, Elisabeth" uniqKey="Diot E" first="Elisabeth" last="Diot">Elisabeth Diot</name>
<affiliation wicri:level="1">
<nlm:aff id="af27">
<addr-line>Service de Médecine Interne</addr-line>
,
<institution>Hôpital Bretonneau Tours</institution>
<addr-line>Cedex</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Madhok, Rajan" sort="Madhok, Rajan" uniqKey="Madhok R" first="Rajan" last="Madhok">Rajan Madhok</name>
<affiliation wicri:level="1">
<nlm:aff id="af28">
<institution>Centre for Rheumatic Diseases</institution>
,
<addr-line>Glasgow Royal Infirmary, Glasgow</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Akil, Mohammed" sort="Akil, Mohammed" uniqKey="Akil M" first="Mohammed" last="Akil">Mohammed Akil</name>
<affiliation wicri:level="1">
<nlm:aff id="af29">
<institution>Sheffield Teaching Hospitals</institution>
,
<addr-line>Sheffield</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Buch, Maya H" sort="Buch, Maya H" uniqKey="Buch M" first="Maya H" last="Buch">Maya H. Buch</name>
<affiliation wicri:level="1">
<nlm:aff id="af30">
<institution>Leeds Institute of Rheumatic and Musculoskeletal Medicine, University of Leeds and NIHR Leeds Musculoskeletal Biomedical Research Unit, Leeds Teaching Hospitals NHS Trust</institution>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Chung, Lorinda" sort="Chung, Lorinda" uniqKey="Chung L" first="Lorinda" last="Chung">Lorinda Chung</name>
<affiliation wicri:level="1">
<nlm:aff id="af31">
<institution>Stanford University</institution>
,
<addr-line>Stanford, California</addr-line>
,
<country>USA</country>
.</nlm:aff>
<country xml:lang="fr">États-Unis</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Damjanov, Nemanja" sort="Damjanov, Nemanja" uniqKey="Damjanov N" first="Nemanja" last="Damjanov">Nemanja Damjanov</name>
<affiliation wicri:level="1">
<nlm:aff id="af32">
<institution>University of Belgrade School of Medicine, Institute of Rheumatology</institution>
,
<addr-line>Belgrade</addr-line>
,
<country>Serbia</country>
</nlm:aff>
<country xml:lang="fr">Serbie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Gunawardena, Harsha" sort="Gunawardena, Harsha" uniqKey="Gunawardena H" first="Harsha" last="Gunawardena">Harsha Gunawardena</name>
<affiliation wicri:level="1">
<nlm:aff id="af33">
<institution>Clinical and Academic Rheumatology, North Bristol NHS Trust</institution>
,
<addr-line>Bristol</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Lanyon, Peter" sort="Lanyon, Peter" uniqKey="Lanyon P" first="Peter" last="Lanyon">Peter Lanyon</name>
<affiliation wicri:level="1">
<nlm:aff id="af34">
<institution>Nottingham University Hospitals NHS Trust, and Nottingham NHS Treatment Centre</institution>
,
<addr-line>Nottingham</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Ahmad, Yasmeen" sort="Ahmad, Yasmeen" uniqKey="Ahmad Y" first="Yasmeen" last="Ahmad">Yasmeen Ahmad</name>
<affiliation wicri:level="1">
<nlm:aff id="af35">
<institution>Peter Maddison Rheumatology Centre</institution>
,
<addr-line>Llandudno</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Chakravarty, Kuntal" sort="Chakravarty, Kuntal" uniqKey="Chakravarty K" first="Kuntal" last="Chakravarty">Kuntal Chakravarty</name>
<affiliation wicri:level="1">
<nlm:aff id="af36">
<institution>Queens Hospital</institution>
,
<addr-line>Romford</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Jacobsen, S Ren" sort="Jacobsen, S Ren" uniqKey="Jacobsen S" first="S Ren" last="Jacobsen">S Ren Jacobsen</name>
<affiliation wicri:level="1">
<nlm:aff id="af37">
<institution>University of Copenhagen, Copenhagen Lupus and Vasculitis Clinic, Center for Rheumatology and Spine Diseases, Rigshospitalet</institution>
,
<addr-line>Copenhagen</addr-line>
,
<country>Denmark</country>
</nlm:aff>
<country xml:lang="fr">Danemark</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Macgregor, Alexander J" sort="Macgregor, Alexander J" uniqKey="Macgregor A" first="Alexander J" last="Macgregor">Alexander J. Macgregor</name>
<affiliation wicri:level="1">
<nlm:aff id="af38">
<institution>Norwich Medical School, University of East Anglia</institution>
,
<addr-line>Norwich</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Mchugh, Neil" sort="Mchugh, Neil" uniqKey="Mchugh N" first="Neil" last="Mchugh">Neil Mchugh</name>
<affiliation wicri:level="1">
<nlm:aff id="af39">
<institution>Royal National Hospital for Rheumatic Diseases</institution>
,
<addr-line>Bath</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Muller Ladner, Ulf" sort="Muller Ladner, Ulf" uniqKey="Muller Ladner U" first="Ulf" last="Müller-Ladner">Ulf Müller-Ladner</name>
<affiliation wicri:level="1">
<nlm:aff id="af40">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>Justus-Liebig University Giessen</institution>
,
<addr-line>Bad Nauheim</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Riemekasten, Gabriela" sort="Riemekasten, Gabriela" uniqKey="Riemekasten G" first="Gabriela" last="Riemekasten">Gabriela Riemekasten</name>
<affiliation wicri:level="1">
<nlm:aff id="af41">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>University of Lübeck</institution>
,
<addr-line>Lübeck</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Becker, Michael" sort="Becker, Michael" uniqKey="Becker M" first="Michael" last="Becker">Michael Becker</name>
<affiliation wicri:level="1">
<nlm:aff id="af42">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>University Hospital Charité Berlin</institution>
,
<addr-line>Berlin</addr-line>
,
<country>Germany</country>
</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Roddy, Janet" sort="Roddy, Janet" uniqKey="Roddy J" first="Janet" last="Roddy">Janet Roddy</name>
<affiliation wicri:level="1">
<nlm:aff id="af43">
<addr-line>Department of Rheumatology</addr-line>
,
<institution>Royal Perth Hospital</institution>
,
<addr-line>Perth</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Carreira, Patricia E" sort="Carreira, Patricia E" uniqKey="Carreira P" first="Patricia E" last="Carreira">Patricia E. Carreira</name>
<affiliation wicri:level="1">
<nlm:aff id="af44">
<institution>Servicio de Reumatologia. Hospital Universitario 12 de Octubre</institution>
,
<addr-line>Madrid</addr-line>
,
<country>Spain</country>
</nlm:aff>
<country xml:lang="fr">Espagne</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Fauchais, Anne Laure" sort="Fauchais, Anne Laure" uniqKey="Fauchais A" first="Anne Laure" last="Fauchais">Anne Laure Fauchais</name>
<affiliation wicri:level="1">
<nlm:aff id="af45">
<addr-line>Internal Medicine Unit</addr-line>
,
<institution>Limoges University Hospital</institution>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hachulla, Eric" sort="Hachulla, Eric" uniqKey="Hachulla E" first="Eric" last="Hachulla">Eric Hachulla</name>
<affiliation wicri:level="1">
<nlm:aff id="af46">
<addr-line>Département de Médecine Interne et Immunologie Clinique</addr-line>
,
<institution>Centre National de Référence Maladies Systémiques etAuto-immunes Rares, Université de Lille, Inserm, U995, FHU Immune-Mediated Inflammatory Diseases and Targeted Therapies,</institution>
<addr-line>Lille</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Hamilton, Jennifer" sort="Hamilton, Jennifer" uniqKey="Hamilton J" first="Jennifer" last="Hamilton">Jennifer Hamilton</name>
<affiliation wicri:level="1">
<nlm:aff id="af47">
<institution>Gateshead Hospitals Foundation Trust</institution>
,
<addr-line>Gateshead</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey=" Nanc, Murat" sort=" Nanc, Murat" uniqKey=" Nanc M" first="Murat" last=" Nanç">Murat Nanç</name>
<affiliation wicri:level="1">
<nlm:aff id="af48">
<addr-line>Department of Internal Medicine, Division of Rheumatology</addr-line>
,
<institution>Istanbul University</institution>
,
<addr-line>Istanbul</addr-line>
,
<country>Turkey</country>
</nlm:aff>
<country xml:lang="fr">Turquie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Mclaren, John S" sort="Mclaren, John S" uniqKey="Mclaren J" first="John S" last="Mclaren">John S. Mclaren</name>
<affiliation wicri:level="1">
<nlm:aff id="af49">
<addr-line>Fife Rheumatic Diseases Unit</addr-line>
,
<institution>Whyteman's Brae Hospital</institution>
,
<addr-line>Kirkcaldy</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Van Laar, Jacob M" sort="Van Laar, Jacob M" uniqKey="Van Laar J" first="Jacob M" last="Van Laar">Jacob M. Van Laar</name>
<affiliation wicri:level="1">
<nlm:aff id="af50">
<addr-line>Department of Rheumatology and Clinical Immunology</addr-line>
,
<institution>UMC Utrecht</institution>
,
<addr-line>Utrecht</addr-line>
,
<country>The Netherlands</country>
</nlm:aff>
<country xml:lang="fr">Pays-Bas</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Pathare, Sanjay" sort="Pathare, Sanjay" uniqKey="Pathare S" first="Sanjay" last="Pathare">Sanjay Pathare</name>
<affiliation wicri:level="1">
<nlm:aff id="af51">
<institution>James Cook University Hospital</institution>
,
<addr-line>Middlesbrough</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Proudman, Susannah" sort="Proudman, Susannah" uniqKey="Proudman S" first="Susannah" last="Proudman">Susannah Proudman</name>
<affiliation>
<nlm:aff id="af52">
<addr-line>Rheumatology Unit</addr-line>
,
<institution>Royal Adelaide Hospital, and Discipline of Medicine, University of Adelaide</institution>
,
<addr-line>Adelaide</addr-line>
,
<country>South Australia</country>
</nlm:aff>
<wicri:noCountry code="nlm country">South Australia</wicri:noCountry>
</affiliation>
</author>
<author>
<name sortKey="Rudin, Anna" sort="Rudin, Anna" uniqKey="Rudin A" first="Anna" last="Rudin">Anna Rudin</name>
<affiliation wicri:level="1">
<nlm:aff id="af53">
<addr-line>Department of Rheumatology and Inflammation Research</addr-line>
,
<institution>The Sahlgrenska Academy at Gothenburg University</institution>
,
<addr-line>Gothenburg</addr-line>
,
<country>Sweden</country>
</nlm:aff>
<country xml:lang="fr">Suède</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Sahhar, Joanne" sort="Sahhar, Joanne" uniqKey="Sahhar J" first="Joanne" last="Sahhar">Joanne Sahhar</name>
<affiliation wicri:level="1">
<nlm:aff id="af54">
<institution>Monash Centre for Inflammatory Diseases, Monash University</institution>
,
<addr-line>Clayton, Melbourne</addr-line>
,
<country>Australia</country>
</nlm:aff>
<country xml:lang="fr">Australie</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Coppere, Brigitte" sort="Coppere, Brigitte" uniqKey="Coppere B" first="Brigitte" last="Coppere">Brigitte Coppere</name>
<affiliation wicri:level="1">
<nlm:aff id="af55">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Hôpital Edouard Herriot</institution>
,
<addr-line>Lyon</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Serratrice, Christine" sort="Serratrice, Christine" uniqKey="Serratrice C" first="Christine" last="Serratrice">Christine Serratrice</name>
<affiliation wicri:level="1">
<nlm:aff id="af56">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Foundation Hospital Saint Joseph</institution>
,
<addr-line>Marseille</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Sheeran, Tom" sort="Sheeran, Tom" uniqKey="Sheeran T" first="Tom" last="Sheeran">Tom Sheeran</name>
<affiliation wicri:level="1">
<nlm:aff id="af57">
<institution>Cannock Chase Hospital</institution>
,
<addr-line>Cannock</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Veale, Douglas J" sort="Veale, Douglas J" uniqKey="Veale D" first="Douglas J" last="Veale">Douglas J. Veale</name>
<affiliation wicri:level="1">
<nlm:aff id="af58">
<institution>St Vincent's University Hospital</institution>
,
<addr-line>Dublin</addr-line>
,
<country>Ireland</country>
</nlm:aff>
<country xml:lang="fr">Irlande (pays)</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Grange, Claire" sort="Grange, Claire" uniqKey="Grange C" first="Claire" last="Grange">Claire Grange</name>
<affiliation wicri:level="1">
<nlm:aff id="af59">
<addr-line>Department of Internal Medicine</addr-line>
,
<institution>Centre Hospitalier Lyon Sud</institution>
,
<addr-line>Pierre Benite</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Trad, Georges Selim" sort="Trad, Georges Selim" uniqKey="Trad G" first="Georges-Selim" last="Trad">Georges-Selim Trad</name>
<affiliation wicri:level="1">
<nlm:aff id="af60">
<addr-line>Internal Medecine</addr-line>
,
<institution>Ambroise Paré Hospital</institution>
,
<addr-line>Boulogne Billancourt</addr-line>
,
<country>France</country>
</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
<author>
<name sortKey="Denton, Christopher P" sort="Denton, Christopher P" uniqKey="Denton C" first="Christopher P" last="Denton">Christopher P. Denton</name>
<affiliation wicri:level="1">
<nlm:aff id="af15">
<addr-line>UCL Division of Medicine</addr-line>
,
<institution>Centre for Rheumatology and Connective Tissue Diseases</institution>
,
<addr-line>London</addr-line>
,
<country>UK</country>
</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea># see nlm:aff country strict</wicri:regionArea>
</affiliation>
</author>
</analytic>
<series>
<title level="j">Annals of the Rheumatic Diseases</title>
<idno type="ISSN">0003-4967</idno>
<idno type="eISSN">1468-2060</idno>
<imprint>
<date when="2017">2017</date>
</imprint>
</series>
</biblStruct>
</sourceDesc>
</fileDesc>
<profileDesc>
<textClass>
<keywords scheme="KwdEn" xml:lang="en">
<term>Adult</term>
<term>Antibodies, Antinuclear (immunology)</term>
<term>Autoantibodies (immunology)</term>
<term>Cohort Studies</term>
<term>Cyclophosphamide (therapeutic use)</term>
<term>Early Medical Intervention</term>
<term>Europe</term>
<term>Female</term>
<term>Humans</term>
<term>Immunosuppressive Agents (therapeutic use)</term>
<term>Male</term>
<term>Methotrexate (therapeutic use)</term>
<term>Middle Aged</term>
<term>Mycophenolic Acid (therapeutic use)</term>
<term>Nuclear Proteins (immunology)</term>
<term>Prospective Studies</term>
<term>RNA Polymerase III (immunology)</term>
<term>Scleroderma, Diffuse (drug therapy)</term>
<term>Scleroderma, Diffuse (immunology)</term>
<term>Severity of Illness Index</term>
<term>Survival Rate</term>
<term>Treatment Outcome</term>
</keywords>
<keywords scheme="KwdFr" xml:lang="fr">
<term>Acide mycophénolique (usage thérapeutique)</term>
<term>Adulte</term>
<term>Adulte d'âge moyen</term>
<term>Anticorps antinucléaires (immunologie)</term>
<term>Autoanticorps (immunologie)</term>
<term>Cyclophosphamide (usage thérapeutique)</term>
<term>Europe</term>
<term>Femelle</term>
<term>Humains</term>
<term>Immunosuppresseurs (usage thérapeutique)</term>
<term>Indice de gravité médicale</term>
<term>Intervention médicale précoce</term>
<term>Mâle</term>
<term>Méthotrexate (usage thérapeutique)</term>
<term>Protéines nucléaires (immunologie)</term>
<term>RNA polymerase III (immunologie)</term>
<term>Résultat thérapeutique</term>
<term>Sclérodermie diffuse (immunologie)</term>
<term>Sclérodermie diffuse (traitement médicamenteux)</term>
<term>Taux de survie</term>
<term>Études de cohortes</term>
<term>Études prospectives</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="immunology" xml:lang="en">
<term>Antibodies, Antinuclear</term>
<term>Autoantibodies</term>
<term>Nuclear Proteins</term>
<term>RNA Polymerase III</term>
</keywords>
<keywords scheme="MESH" type="chemical" qualifier="therapeutic use" xml:lang="en">
<term>Cyclophosphamide</term>
<term>Immunosuppressive Agents</term>
<term>Methotrexate</term>
<term>Mycophenolic Acid</term>
</keywords>
<keywords scheme="MESH" qualifier="drug therapy" xml:lang="en">
<term>Scleroderma, Diffuse</term>
</keywords>
<keywords scheme="MESH" qualifier="immunologie" xml:lang="fr">
<term>Anticorps antinucléaires</term>
<term>Autoanticorps</term>
<term>Protéines nucléaires</term>
<term>RNA polymerase III</term>
<term>Sclérodermie diffuse</term>
</keywords>
<keywords scheme="MESH" qualifier="immunology" xml:lang="en">
<term>Scleroderma, Diffuse</term>
</keywords>
<keywords scheme="MESH" qualifier="traitement médicamenteux" xml:lang="fr">
<term>Sclérodermie diffuse</term>
</keywords>
<keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr">
<term>Acide mycophénolique</term>
<term>Cyclophosphamide</term>
<term>Immunosuppresseurs</term>
<term>Méthotrexate</term>
</keywords>
<keywords scheme="MESH" xml:lang="en">
<term>Adult</term>
<term>Cohort Studies</term>
<term>Early Medical Intervention</term>
<term>Europe</term>
<term>Female</term>
<term>Humans</term>
<term>Male</term>
<term>Middle Aged</term>
<term>Prospective Studies</term>
<term>Severity of Illness Index</term>
<term>Survival Rate</term>
<term>Treatment Outcome</term>
</keywords>
<keywords scheme="MESH" xml:lang="fr">
<term>Adulte</term>
<term>Adulte d'âge moyen</term>
<term>Europe</term>
<term>Femelle</term>
<term>Humains</term>
<term>Indice de gravité médicale</term>
<term>Intervention médicale précoce</term>
<term>Mâle</term>
<term>Résultat thérapeutique</term>
<term>Taux de survie</term>
<term>Études de cohortes</term>
<term>Études prospectives</term>
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</teiHeader>
<front>
<div type="abstract" xml:lang="en">
<sec>
<title>Objectives</title>
<p>The rarity of early diffuse cutaneous systemic sclerosis (dcSSc) makes randomised controlled trials very difficult. We aimed to use an observational approach to compare effectiveness of currently used treatment approaches.</p>
</sec>
<sec>
<title>Methods</title>
<p>This was a prospective, observational cohort study of early dcSSc (within three years of onset of skin thickening). Clinicians selected one of four protocols for each patient: methotrexate, mycophenolate mofetil (MMF), cyclophosphamide or ‘no immunosuppressant’. Patients were assessed three-monthly for up to 24 months. The primary outcome was the change in modified Rodnan skin score (mRSS). Confounding by indication at baseline was accounted for using inverse probability of treatment (IPT) weights. As a secondary outcome, an IPT-weighted Cox model was used to test for differences in survival.</p>
</sec>
<sec>
<title>Results</title>
<p>Of 326 patients recruited from 50 centres, 65 were prescribed methotrexate, 118 MMF, 87 cyclophosphamide and 56 no immunosuppressant. 276 (84.7%) patients completed 12 and 234 (71.7%) 24 months follow-up (or reached last visit date). There were statistically significant reductions in mRSS at 12 months in all groups: −4.0 (−5.2 to −2.7) units for methotrexate, −4.1 (−5.3 to −2.9) for MMF, −3.3 (−4.9 to −1.7) for cyclophosphamide and −2.2 (−4.0 to −0.3) for no immunosuppressant (p value for between-group differences=0.346). There were no statistically significant differences in survival between protocols before (p=0.389) or after weighting (p=0.440), but survival was poorest in the no immunosuppressant group (84.0%) at 24 months.</p>
</sec>
<sec>
<title>Conclusions</title>
<p>These findings may support using immunosuppressants for early dcSSc but suggest that overall benefit is modest over 12 months and that better treatments are needed.</p>
</sec>
<sec>
<title>Trial registration number</title>
<p>NCT02339441.</p>
</sec>
</div>
</front>
<back>
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