The Paediatric Rheumatology International Trials Organisation Provisional Criteria for the Evaluation of Response to Therapy in Juvenile Dermatomyositis
Identifieur interne : 003D67 ( PascalFrancis/Curation ); précédent : 003D66; suivant : 003D68The Paediatric Rheumatology International Trials Organisation Provisional Criteria for the Evaluation of Response to Therapy in Juvenile Dermatomyositis
Auteurs : Nicolino Ruperto [Italie] ; Angela Pistorio [Italie] ; Angelo Ravelli [Italie] ; Lisa G. Rider [États-Unis] ; Clarissa Pilkington [Royaume-Uni] ; Sheila Oliveira [Brésil] ; Nico Wulffraat [Pays-Bas] ; Graciela Espada [Argentine] ; Stella Garay [Argentine] ; Ruben Cuttica [Argentine] ; Michael Hofer [Suisse] ; Pierre Quartier [France] ; Jose Melo-Gomes [Portugal] ; Ann M. Reed [États-Unis] ; Malgorzata Wierzbowska [Pologne] ; Brian M. Feldman [Canada] ; Miroslav Harjacek [Croatie] ; Hans-Iko Huppertz [Allemagne] ; Susan Nielsen [Danemark] ; Berit Flato [Norvège] ; Pekka Lahdenne [Finlande] ; Harmut Michels [Allemagne] ; Kevin J. Murray [Australie] ; Lynn Punaro [États-Unis] ; Robert Rennebohm [Canada] ; Ricardo Russo ; Zsolt Balogh ; Madeleine Rooney ; Lauren M. Pachman ; Carol Wallace ; Philip Hashkes ; Daniel J. Lovell ; Edward H. Giannini ; Boel Andersson Gare ; Alberto Martini [Italie]Source :
- Arthritis care and research [ 0893-7524 ] ; 2010.
Descripteurs français
- Pascal (Inist)
- Wicri :
- topic : Enfant.
English descriptors
- KwdEn :
Abstract
Objective. To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (DM) based on the Paediatric Rheumatology International Trials Organisation juvenile DM core set of variables. Methods. Thirty-seven experienced pediatric rheumatologists from 27 countries achieved consensus on 128 difficult patient profiles as clinically improved or not improved using a stepwise approach (patient's rating, statistical analysis, definition selection). Using the physicians' consensus ratings as the "gold standard measure," chi-square, sensitivity, specificity, false-positive and-negative rates, area under the receiver operating characteristic curve, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa values >0.8 were multiplied by the face validity score to select the top definitions. Results. The top definition of improvement was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 1 of the remaining worsening by more than 30%, which cannot be muscle strength. The second-highest scoring definition was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength (definition P1 selected by the International Myositis Assessment and Clinical Studies group). The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process. Conclusion. We propose a provisional data-driven definition of improvement that reflects well the consensus rating of experienced clinicians, which incorporates clinically meaningful change in core set variables in a composite end point for the evaluation of global response to therapy in juvenile DM.
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<author><name sortKey="Punaro, Lynn" sort="Punaro, Lynn" uniqKey="Punaro L" first="Lynn" last="Punaro">Lynn Punaro</name>
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<author><name sortKey="Pachman, Lauren M" sort="Pachman, Lauren M" uniqKey="Pachman L" first="Lauren M." last="Pachman">Lauren M. Pachman</name>
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<author><name sortKey="Wallace, Carol" sort="Wallace, Carol" uniqKey="Wallace C" first="Carol" last="Wallace">Carol Wallace</name>
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<author><name sortKey="Hashkes, Philip" sort="Hashkes, Philip" uniqKey="Hashkes P" first="Philip" last="Hashkes">Philip Hashkes</name>
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<author><name sortKey="Lovell, Daniel J" sort="Lovell, Daniel J" uniqKey="Lovell D" first="Daniel J." last="Lovell">Daniel J. Lovell</name>
</author>
<author><name sortKey="Giannini, Edward H" sort="Giannini, Edward H" uniqKey="Giannini E" first="Edward H." last="Giannini">Edward H. Giannini</name>
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<author><name sortKey="Andersson Gare, Boel" sort="Andersson Gare, Boel" uniqKey="Andersson Gare B" first="Boel" last="Andersson Gare">Boel Andersson Gare</name>
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<author><name sortKey="Martini, Alberto" sort="Martini, Alberto" uniqKey="Martini A" first="Alberto" last="Martini">Alberto Martini</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en" level="a">The Paediatric Rheumatology International Trials Organisation Provisional Criteria for the Evaluation of Response to Therapy in Juvenile Dermatomyositis</title>
<author><name sortKey="Ruperto, Nicolino" sort="Ruperto, Nicolino" uniqKey="Ruperto N" first="Nicolino" last="Ruperto">Nicolino Ruperto</name>
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<author><name sortKey="Pistorio, Angela" sort="Pistorio, Angela" uniqKey="Pistorio A" first="Angela" last="Pistorio">Angela Pistorio</name>
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<author><name sortKey="Ravelli, Angelo" sort="Ravelli, Angelo" uniqKey="Ravelli A" first="Angelo" last="Ravelli">Angelo Ravelli</name>
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<author><name sortKey="Rider, Lisa G" sort="Rider, Lisa G" uniqKey="Rider L" first="Lisa G." last="Rider">Lisa G. Rider</name>
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<author><name sortKey="Pilkington, Clarissa" sort="Pilkington, Clarissa" uniqKey="Pilkington C" first="Clarissa" last="Pilkington">Clarissa Pilkington</name>
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<author><name sortKey="Oliveira, Sheila" sort="Oliveira, Sheila" uniqKey="Oliveira S" first="Sheila" last="Oliveira">Sheila Oliveira</name>
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<s2>Rio de Janeiro</s2>
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<author><name sortKey="Wulffraat, Nico" sort="Wulffraat, Nico" uniqKey="Wulffraat N" first="Nico" last="Wulffraat">Nico Wulffraat</name>
<affiliation wicri:level="1"><inist:fA14 i1="07"><s1>Wilhelmina Kinderziekenhuis</s1>
<s2>Utrecht</s2>
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<author><name sortKey="Espada, Graciela" sort="Espada, Graciela" uniqKey="Espada G" first="Graciela" last="Espada">Graciela Espada</name>
<affiliation wicri:level="1"><inist:fA14 i1="08"><s1>Hospital de Ninos Ricardo Gutierrez</s1>
<s2>Buenos Aires</s2>
<s3>ARG</s3>
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<country>Argentine</country>
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<author><name sortKey="Garay, Stella" sort="Garay, Stella" uniqKey="Garay S" first="Stella" last="Garay">Stella Garay</name>
<affiliation wicri:level="1"><inist:fA14 i1="09"><s1>Hospital Sor Maria Ludovica</s1>
<s2>La Plata</s2>
<s3>ARG</s3>
<sZ>9 aut.</sZ>
</inist:fA14>
<country>Argentine</country>
</affiliation>
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<author><name sortKey="Cuttica, Ruben" sort="Cuttica, Ruben" uniqKey="Cuttica R" first="Ruben" last="Cuttica">Ruben Cuttica</name>
<affiliation wicri:level="1"><inist:fA14 i1="10"><s1>Hospital General de Ninos Pedro de Elizalde</s1>
<s2>Buenos Aires</s2>
<s3>ARG</s3>
<sZ>10 aut.</sZ>
</inist:fA14>
<country>Argentine</country>
</affiliation>
</author>
<author><name sortKey="Hofer, Michael" sort="Hofer, Michael" uniqKey="Hofer M" first="Michael" last="Hofer">Michael Hofer</name>
<affiliation wicri:level="1"><inist:fA14 i1="11"><s1>Centre Multisite Romand de Rhumatologie Pediatrique</s1>
<s2>Lausanne</s2>
<s3>CHE</s3>
<sZ>11 aut.</sZ>
</inist:fA14>
<country>Suisse</country>
</affiliation>
</author>
<author><name sortKey="Quartier, Pierre" sort="Quartier, Pierre" uniqKey="Quartier P" first="Pierre" last="Quartier">Pierre Quartier</name>
<affiliation wicri:level="1"><inist:fA14 i1="12"><s1>Universite Paris-Descartes, Hopital Necker-Enfants Malades</s1>
<s2>Paris</s2>
<s3>FRA</s3>
<sZ>12 aut.</sZ>
</inist:fA14>
<country>France</country>
</affiliation>
</author>
<author><name sortKey="Melo Gomes, Jose" sort="Melo Gomes, Jose" uniqKey="Melo Gomes J" first="Jose" last="Melo-Gomes">Jose Melo-Gomes</name>
<affiliation wicri:level="1"><inist:fA14 i1="13"><s1>Instituto Portugues de Reumatologia</s1>
<s2>Lisbon</s2>
<s3>PRT</s3>
<sZ>13 aut.</sZ>
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<country>Portugal</country>
</affiliation>
</author>
<author><name sortKey="Reed, Ann M" sort="Reed, Ann M" uniqKey="Reed A" first="Ann M." last="Reed">Ann M. Reed</name>
<affiliation wicri:level="1"><inist:fA14 i1="14"><s1>Mayo Clinic School of Medicine and Mayo Foundation</s1>
<s2>Rochester, Minnesota</s2>
<s3>USA</s3>
<sZ>14 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
</affiliation>
</author>
<author><name sortKey="Wierzbowska, Malgorzata" sort="Wierzbowska, Malgorzata" uniqKey="Wierzbowska M" first="Malgorzata" last="Wierzbowska">Malgorzata Wierzbowska</name>
<affiliation wicri:level="1"><inist:fA14 i1="15"><s1>Institute of Rheumatology</s1>
<s2>Warsaw</s2>
<s3>POL</s3>
<sZ>15 aut.</sZ>
</inist:fA14>
<country>Pologne</country>
</affiliation>
</author>
<author><name sortKey="Feldman, Brian M" sort="Feldman, Brian M" uniqKey="Feldman B" first="Brian M." last="Feldman">Brian M. Feldman</name>
<affiliation wicri:level="1"><inist:fA14 i1="16"><s1>University of Toronto and The Hospital for Sick Children</s1>
<s2>Toronto, Ontario</s2>
<s3>CAN</s3>
<sZ>16 aut.</sZ>
</inist:fA14>
<country>Canada</country>
</affiliation>
</author>
<author><name sortKey="Harjacek, Miroslav" sort="Harjacek, Miroslav" uniqKey="Harjacek M" first="Miroslav" last="Harjacek">Miroslav Harjacek</name>
<affiliation wicri:level="1"><inist:fA14 i1="17"><s1>Children's Hospital Zagreb</s1>
<s2>Zagreb</s2>
<s3>HRV</s3>
<sZ>17 aut.</sZ>
</inist:fA14>
<country>Croatie</country>
</affiliation>
</author>
<author><name sortKey="Huppertz, Hans Iko" sort="Huppertz, Hans Iko" uniqKey="Huppertz H" first="Hans-Iko" last="Huppertz">Hans-Iko Huppertz</name>
<affiliation wicri:level="1"><inist:fA14 i1="18"><s1>Klinikum Bremen-Mitte</s1>
<s2>Bremen</s2>
<s3>DEU</s3>
<sZ>18 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
</affiliation>
</author>
<author><name sortKey="Nielsen, Susan" sort="Nielsen, Susan" uniqKey="Nielsen S" first="Susan" last="Nielsen">Susan Nielsen</name>
<affiliation wicri:level="1"><inist:fA14 i1="19"><s1>Juliane Marie Centret, Rigshospitalet</s1>
<s2>Copenhagen</s2>
<s3>DNK</s3>
<sZ>19 aut.</sZ>
</inist:fA14>
<country>Danemark</country>
</affiliation>
</author>
<author><name sortKey="Flato, Berit" sort="Flato, Berit" uniqKey="Flato B" first="Berit" last="Flato">Berit Flato</name>
<affiliation wicri:level="1"><inist:fA14 i1="20"><s1>Rikshospitalet University Hospital</s1>
<s2>Oslo</s2>
<s3>NOR</s3>
<sZ>20 aut.</sZ>
</inist:fA14>
<country>Norvège</country>
</affiliation>
</author>
<author><name sortKey="Lahdenne, Pekka" sort="Lahdenne, Pekka" uniqKey="Lahdenne P" first="Pekka" last="Lahdenne">Pekka Lahdenne</name>
<affiliation wicri:level="1"><inist:fA14 i1="21"><s1>Hospital for Children and Adolescents</s1>
<s2>Helsinki</s2>
<s3>FIN</s3>
<sZ>21 aut.</sZ>
</inist:fA14>
<country>Finlande</country>
</affiliation>
</author>
<author><name sortKey="Michels, Harmut" sort="Michels, Harmut" uniqKey="Michels H" first="Harmut" last="Michels">Harmut Michels</name>
<affiliation wicri:level="1"><inist:fA14 i1="22"><s1>Rheumakinderklinik</s1>
<s2>Garmisch-Parten-kirchen</s2>
<s3>DEU</s3>
<sZ>22 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
</affiliation>
</author>
<author><name sortKey="Murray, Kevin J" sort="Murray, Kevin J" uniqKey="Murray K" first="Kevin J." last="Murray">Kevin J. Murray</name>
<affiliation wicri:level="1"><inist:fA14 i1="23"><s1>Princess Margaret Hospital for Children</s1>
<s2>Perth, Western Australia</s2>
<s3>AUS</s3>
<sZ>23 aut.</sZ>
</inist:fA14>
<country>Australie</country>
</affiliation>
</author>
<author><name sortKey="Punaro, Lynn" sort="Punaro, Lynn" uniqKey="Punaro L" first="Lynn" last="Punaro">Lynn Punaro</name>
<affiliation wicri:level="1"><inist:fA14 i1="24"><s1>Texas Scottish Rite Hospital</s1>
<s2>Dallas</s2>
<s3>USA</s3>
<sZ>24 aut.</sZ>
</inist:fA14>
<country>États-Unis</country>
</affiliation>
</author>
<author><name sortKey="Rennebohm, Robert" sort="Rennebohm, Robert" uniqKey="Rennebohm R" first="Robert" last="Rennebohm">Robert Rennebohm</name>
<affiliation wicri:level="1"><inist:fA14 i1="25"><s1>Alberta Children's Hospital</s1>
<s2>Calgary, Alberta</s2>
<s3>CAN</s3>
<sZ>25 aut.</sZ>
</inist:fA14>
<country>Canada</country>
</affiliation>
</author>
<author><name sortKey="Russo, Ricardo" sort="Russo, Ricardo" uniqKey="Russo R" first="Ricardo" last="Russo">Ricardo Russo</name>
</author>
<author><name sortKey="Balogh, Zsolt" sort="Balogh, Zsolt" uniqKey="Balogh Z" first="Zsolt" last="Balogh">Zsolt Balogh</name>
</author>
<author><name sortKey="Rooney, Madeleine" sort="Rooney, Madeleine" uniqKey="Rooney M" first="Madeleine" last="Rooney">Madeleine Rooney</name>
</author>
<author><name sortKey="Pachman, Lauren M" sort="Pachman, Lauren M" uniqKey="Pachman L" first="Lauren M." last="Pachman">Lauren M. Pachman</name>
</author>
<author><name sortKey="Wallace, Carol" sort="Wallace, Carol" uniqKey="Wallace C" first="Carol" last="Wallace">Carol Wallace</name>
</author>
<author><name sortKey="Hashkes, Philip" sort="Hashkes, Philip" uniqKey="Hashkes P" first="Philip" last="Hashkes">Philip Hashkes</name>
</author>
<author><name sortKey="Lovell, Daniel J" sort="Lovell, Daniel J" uniqKey="Lovell D" first="Daniel J." last="Lovell">Daniel J. Lovell</name>
</author>
<author><name sortKey="Giannini, Edward H" sort="Giannini, Edward H" uniqKey="Giannini E" first="Edward H." last="Giannini">Edward H. Giannini</name>
</author>
<author><name sortKey="Andersson Gare, Boel" sort="Andersson Gare, Boel" uniqKey="Andersson Gare B" first="Boel" last="Andersson Gare">Boel Andersson Gare</name>
</author>
<author><name sortKey="Martini, Alberto" sort="Martini, Alberto" uniqKey="Martini A" first="Alberto" last="Martini">Alberto Martini</name>
<affiliation wicri:level="1"><inist:fA14 i1="03"><s1>IRCCS G. Gaslini, Pediatria II, Reumatologia, and Università degli Studi</s1>
<s2>Genoa</s2>
<s3>ITA</s3>
<sZ>3 aut.</sZ>
<sZ>35 aut.</sZ>
</inist:fA14>
<country>Italie</country>
</affiliation>
</author>
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<series><title level="j" type="main">Arthritis care and research</title>
<title level="j" type="abbreviated">Arthritis care res.</title>
<idno type="ISSN">0893-7524</idno>
<imprint><date when="2010">2010</date>
</imprint>
</series>
</biblStruct>
</sourceDesc>
<seriesStmt><title level="j" type="main">Arthritis care and research</title>
<title level="j" type="abbreviated">Arthritis care res.</title>
<idno type="ISSN">0893-7524</idno>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term>
<term>Child</term>
<term>Dermatomyositis</term>
<term>Rheumatology</term>
<term>Treatment</term>
</keywords>
<keywords scheme="Pascal" xml:lang="fr"><term>Rhumatologie</term>
<term>Traitement</term>
<term>Dermatomyosite</term>
<term>Enfant</term>
<term>Adolescent</term>
</keywords>
<keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Enfant</term>
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<front><div type="abstract" xml:lang="en">Objective. To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (DM) based on the Paediatric Rheumatology International Trials Organisation juvenile DM core set of variables. Methods. Thirty-seven experienced pediatric rheumatologists from 27 countries achieved consensus on 128 difficult patient profiles as clinically improved or not improved using a stepwise approach (patient's rating, statistical analysis, definition selection). Using the physicians' consensus ratings as the "gold standard measure," chi-square, sensitivity, specificity, false-positive and-negative rates, area under the receiver operating characteristic curve, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa values >0.8 were multiplied by the face validity score to select the top definitions. Results. The top definition of improvement was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 1 of the remaining worsening by more than 30%, which cannot be muscle strength. The second-highest scoring definition was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength (definition P1 selected by the International Myositis Assessment and Clinical Studies group). The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process. Conclusion. We propose a provisional data-driven definition of improvement that reflects well the consensus rating of experienced clinicians, which incorporates clinically meaningful change in core set variables in a composite end point for the evaluation of global response to therapy in juvenile DM.</div>
</front>
</TEI>
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<fA08 i1="01" i2="1" l="ENG"><s1>The Paediatric Rheumatology International Trials Organisation Provisional Criteria for the Evaluation of Response to Therapy in Juvenile Dermatomyositis</s1>
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<fA11 i1="01" i2="1"><s1>RUPERTO (Nicolino)</s1>
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<fA11 i1="18" i2="1"><s1>HUPPERTZ (Hans-Iko)</s1>
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<fA11 i1="21" i2="1"><s1>LAHDENNE (Pekka)</s1>
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<fA11 i1="22" i2="1"><s1>MICHELS (Harmut)</s1>
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<fA11 i1="23" i2="1"><s1>MURRAY (Kevin J.)</s1>
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<fA11 i1="24" i2="1"><s1>PUNARO (Lynn)</s1>
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<fA11 i1="25" i2="1"><s1>RENNEBOHM (Robert)</s1>
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<fA11 i1="26" i2="1"><s1>RUSSO (Ricardo)</s1>
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<fA11 i1="27" i2="1"><s1>BALOGH (Zsolt)</s1>
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<fA11 i1="28" i2="1"><s1>ROONEY (Madeleine)</s1>
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<fA11 i1="29" i2="1"><s1>PACHMAN (Lauren M.)</s1>
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<fA11 i1="30" i2="1"><s1>WALLACE (Carol)</s1>
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<fA11 i1="31" i2="1"><s1>HASHKES (Philip)</s1>
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<fA11 i1="32" i2="1"><s1>LOVELL (Daniel J.)</s1>
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<fA11 i1="33" i2="1"><s1>GIANNINI (Edward H.)</s1>
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<fA11 i1="34" i2="1"><s1>ANDERSSON GARE (Boel)</s1>
</fA11>
<fA11 i1="35" i2="1"><s1>MARTINI (Alberto)</s1>
</fA11>
<fA14 i1="01"><s1>IRCCS G. Gaslini, Pediatria II, Reumatologia, and Paediatric Rheumatology International Trials Organisation</s1>
<s2>Genoa</s2>
<s3>ITA</s3>
<sZ>1 aut.</sZ>
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<fA14 i1="02"><s1>IRCCS G. Gaslini, Servizio di Epidemiologia e Biostatistica</s1>
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<sZ>2 aut.</sZ>
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<fA14 i1="03"><s1>IRCCS G. Gaslini, Pediatria II, Reumatologia, and Università degli Studi</s1>
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<s3>ITA</s3>
<sZ>3 aut.</sZ>
<sZ>35 aut.</sZ>
</fA14>
<fA14 i1="04"><s1>National Institute of Environmental Health Sciences, NIH</s1>
<s2>Bethesda, Maryland</s2>
<s3>USA</s3>
<sZ>4 aut.</sZ>
</fA14>
<fA14 i1="05"><s1>Great Ormond Street Hospital for Sick Children</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>5 aut.</sZ>
</fA14>
<fA14 i1="06"><s1>Universidade Federal do Rio de Janeiro, Instituto de Puericultura e Pediatria Martagao Gesteira</s1>
<s2>Rio de Janeiro</s2>
<s3>BRA</s3>
<sZ>6 aut.</sZ>
</fA14>
<fA14 i1="07"><s1>Wilhelmina Kinderziekenhuis</s1>
<s2>Utrecht</s2>
<s3>NLD</s3>
<sZ>7 aut.</sZ>
</fA14>
<fA14 i1="08"><s1>Hospital de Ninos Ricardo Gutierrez</s1>
<s2>Buenos Aires</s2>
<s3>ARG</s3>
<sZ>8 aut.</sZ>
</fA14>
<fA14 i1="09"><s1>Hospital Sor Maria Ludovica</s1>
<s2>La Plata</s2>
<s3>ARG</s3>
<sZ>9 aut.</sZ>
</fA14>
<fA14 i1="10"><s1>Hospital General de Ninos Pedro de Elizalde</s1>
<s2>Buenos Aires</s2>
<s3>ARG</s3>
<sZ>10 aut.</sZ>
</fA14>
<fA14 i1="11"><s1>Centre Multisite Romand de Rhumatologie Pediatrique</s1>
<s2>Lausanne</s2>
<s3>CHE</s3>
<sZ>11 aut.</sZ>
</fA14>
<fA14 i1="12"><s1>Universite Paris-Descartes, Hopital Necker-Enfants Malades</s1>
<s2>Paris</s2>
<s3>FRA</s3>
<sZ>12 aut.</sZ>
</fA14>
<fA14 i1="13"><s1>Instituto Portugues de Reumatologia</s1>
<s2>Lisbon</s2>
<s3>PRT</s3>
<sZ>13 aut.</sZ>
</fA14>
<fA14 i1="14"><s1>Mayo Clinic School of Medicine and Mayo Foundation</s1>
<s2>Rochester, Minnesota</s2>
<s3>USA</s3>
<sZ>14 aut.</sZ>
</fA14>
<fA14 i1="15"><s1>Institute of Rheumatology</s1>
<s2>Warsaw</s2>
<s3>POL</s3>
<sZ>15 aut.</sZ>
</fA14>
<fA14 i1="16"><s1>University of Toronto and The Hospital for Sick Children</s1>
<s2>Toronto, Ontario</s2>
<s3>CAN</s3>
<sZ>16 aut.</sZ>
</fA14>
<fA14 i1="17"><s1>Children's Hospital Zagreb</s1>
<s2>Zagreb</s2>
<s3>HRV</s3>
<sZ>17 aut.</sZ>
</fA14>
<fA14 i1="18"><s1>Klinikum Bremen-Mitte</s1>
<s2>Bremen</s2>
<s3>DEU</s3>
<sZ>18 aut.</sZ>
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<fA14 i1="19"><s1>Juliane Marie Centret, Rigshospitalet</s1>
<s2>Copenhagen</s2>
<s3>DNK</s3>
<sZ>19 aut.</sZ>
</fA14>
<fA14 i1="20"><s1>Rikshospitalet University Hospital</s1>
<s2>Oslo</s2>
<s3>NOR</s3>
<sZ>20 aut.</sZ>
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<fA14 i1="21"><s1>Hospital for Children and Adolescents</s1>
<s2>Helsinki</s2>
<s3>FIN</s3>
<sZ>21 aut.</sZ>
</fA14>
<fA14 i1="22"><s1>Rheumakinderklinik</s1>
<s2>Garmisch-Parten-kirchen</s2>
<s3>DEU</s3>
<sZ>22 aut.</sZ>
</fA14>
<fA14 i1="23"><s1>Princess Margaret Hospital for Children</s1>
<s2>Perth, Western Australia</s2>
<s3>AUS</s3>
<sZ>23 aut.</sZ>
</fA14>
<fA14 i1="24"><s1>Texas Scottish Rite Hospital</s1>
<s2>Dallas</s2>
<s3>USA</s3>
<sZ>24 aut.</sZ>
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<fA14 i1="25"><s1>Alberta Children's Hospital</s1>
<s2>Calgary, Alberta</s2>
<s3>CAN</s3>
<sZ>25 aut.</sZ>
</fA14>
<fA17 i1="01" i2="1"><s1>PEDIATRIC RHEUMATOLOGY COLLABORATIVE STUDY GROUP (PRCSG)</s1>
<s3>INC</s3>
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<fA20><s1>1533-1541</s1>
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<fC01 i1="01" l="ENG"><s0>Objective. To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (DM) based on the Paediatric Rheumatology International Trials Organisation juvenile DM core set of variables. Methods. Thirty-seven experienced pediatric rheumatologists from 27 countries achieved consensus on 128 difficult patient profiles as clinically improved or not improved using a stepwise approach (patient's rating, statistical analysis, definition selection). Using the physicians' consensus ratings as the "gold standard measure," chi-square, sensitivity, specificity, false-positive and-negative rates, area under the receiver operating characteristic curve, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa values >0.8 were multiplied by the face validity score to select the top definitions. Results. The top definition of improvement was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 1 of the remaining worsening by more than 30%, which cannot be muscle strength. The second-highest scoring definition was at least 20% improvement from baseline in 3 of 6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength (definition P1 selected by the International Myositis Assessment and Clinical Studies group). The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process. Conclusion. We propose a provisional data-driven definition of improvement that reflects well the consensus rating of experienced clinicians, which incorporates clinically meaningful change in core set variables in a composite end point for the evaluation of global response to therapy in juvenile DM.</s0>
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<s5>10</s5>
</fC03>
<fC03 i1="04" i2="X" l="SPA"><s0>Niño</s0>
<s5>10</s5>
</fC03>
<fC03 i1="05" i2="X" l="FRE"><s0>Adolescent</s0>
<s5>11</s5>
</fC03>
<fC03 i1="05" i2="X" l="ENG"><s0>Adolescent</s0>
<s5>11</s5>
</fC03>
<fC03 i1="05" i2="X" l="SPA"><s0>Adolescente</s0>
<s5>11</s5>
</fC03>
<fC07 i1="01" i2="X" l="FRE"><s0>Homme</s0>
</fC07>
<fC07 i1="01" i2="X" l="ENG"><s0>Human</s0>
</fC07>
<fC07 i1="01" i2="X" l="SPA"><s0>Hombre</s0>
</fC07>
<fC07 i1="02" i2="X" l="FRE"><s0>Maladie autoimmune</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="ENG"><s0>Autoimmune disease</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="SPA"><s0>Enfermedad autoinmune</s0>
<s5>37</s5>
</fC07>
<fC07 i1="03" i2="X" l="FRE"><s0>Maladie de système</s0>
<s5>38</s5>
</fC07>
<fC07 i1="03" i2="X" l="ENG"><s0>Systemic disease</s0>
<s5>38</s5>
</fC07>
<fC07 i1="03" i2="X" l="SPA"><s0>Enfermedad sistémica</s0>
<s5>38</s5>
</fC07>
<fC07 i1="04" i2="X" l="FRE"><s0>Pathologie du muscle strié</s0>
<s5>39</s5>
</fC07>
<fC07 i1="04" i2="X" l="ENG"><s0>Striated muscle disease</s0>
<s5>39</s5>
</fC07>
<fC07 i1="04" i2="X" l="SPA"><s0>Músculo estriado patología</s0>
<s5>39</s5>
</fC07>
<fC07 i1="05" i2="X" l="FRE"><s0>Pathologie de la peau</s0>
<s5>40</s5>
</fC07>
<fC07 i1="05" i2="X" l="ENG"><s0>Skin disease</s0>
<s5>40</s5>
</fC07>
<fC07 i1="05" i2="X" l="SPA"><s0>Piel patología</s0>
<s5>40</s5>
</fC07>
<fC07 i1="06" i2="X" l="FRE"><s0>Pathologie du tissu conjonctif</s0>
<s5>41</s5>
</fC07>
<fC07 i1="06" i2="X" l="ENG"><s0>Connective tissue disease</s0>
<s5>41</s5>
</fC07>
<fC07 i1="06" i2="X" l="SPA"><s0>Tejido conjuntivo patología</s0>
<s5>41</s5>
</fC07>
<fC07 i1="07" i2="X" l="FRE"><s0>Immunopathologie</s0>
<s5>42</s5>
</fC07>
<fC07 i1="07" i2="X" l="ENG"><s0>Immunopathology</s0>
<s5>42</s5>
</fC07>
<fC07 i1="07" i2="X" l="SPA"><s0>Inmunopatología</s0>
<s5>42</s5>
</fC07>
<fN21><s1>003</s1>
</fN21>
<fN44 i1="01"><s1>OTO</s1>
</fN44>
<fN82><s1>OTO</s1>
</fN82>
</pA>
</standard>
</inist>
</record>
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