THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION PROVISIONAL CRITERIA FOR THE EVALUATION OF RESPONSE TO THERAPY IN JUVENILE DERMATOMYOSITIS
Identifieur interne : 001439 ( Pmc/Corpus ); précédent : 001438; suivant : 001440THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION PROVISIONAL CRITERIA FOR THE EVALUATION OF RESPONSE TO THERAPY IN JUVENILE DERMATOMYOSITIS
Auteurs : Nicolino Ruperto ; Angela Pistorio ; Angelo Ravelli ; Lisa G. Rider ; Clarissa Pilkington ; Sheila Oliveira ; Nico Wulffraat ; Graciela Espada ; Stella Garay ; Ruben Cuttica ; Michael Hofer ; Pierre Quartier ; Jose Melo-Gomes ; Ann M. Reed ; Malgorzata Wierzbowska ; Brian M. Feldman ; Miroslav Harjacek ; Hans-Iko Huppertz ; Susan Nielsen ; Berit Flato ; Pekka Lahdenne ; Harmut Michels ; Kevin J. Murray ; Lynn Punaro ; Robert Rennebohm ; Ricardo Russo ; Zsolt Balogh ; Madeleine Rooney ; Lauren M. Pachman ; Carol Wallace ; Philip Hashkes ; Daniel J. Lovell ; Edward H. Giannini ; Alberto MartiniSource :
- Arthritis care & research [ 2151-464X ] ; 2010.
Abstract
To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (JDM) based on the PRINTO JDM core set of variables.
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 (patients rating, statistical analysis, definition selection). Using the physicians’ consensus ratings as the “gold-standard measure”, chi-square, sensitivity, specificity, false positive and negative rate, area under the ROC, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa >0.8 were multiplied with the face validity score to select the top definitions.
The top definition of improvement was: at least 20% improvement from baseline in 3/6 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 3/6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength which is definition P1 selected by the IMACS group. The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process.
we proposes 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 endpoint for the evaluation of global response to therapy in JDM.
Url:
DOI: 10.1002/acr.20280
PubMed: 20583105
PubMed Central: 2964396
Links to Exploration step
PMC:2964396Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION 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><affiliation><nlm:aff id="A1">Nicolino Ruperto, MD, MPH, IRCCS G Gaslini, Pediatria II, Reumatologia, PRINTO, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Pistorio, Angela" sort="Pistorio, Angela" uniqKey="Pistorio A" first="Angela" last="Pistorio">Angela Pistorio</name><affiliation><nlm:aff id="A2">Angela Pistorio, MD, PhD, IRCCS G Gaslini, Servizio di Epidemiologia e Biostatistica, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Ravelli, Angelo" sort="Ravelli, Angelo" uniqKey="Ravelli A" first="Angelo" last="Ravelli">Angelo Ravelli</name><affiliation><nlm:aff id="A1">Nicolino Ruperto, MD, MPH, IRCCS G Gaslini, Pediatria II, Reumatologia, PRINTO, Genova, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="A3">Angelo Ravelli, MD, Prof, Alberto Martini, MD, Prof, IRCCS G. Gaslini, Pediatria II, Reumatologia and Dipartimento di Pediatria, Università degli Studi, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Rider, Lisa G" sort="Rider, Lisa G" uniqKey="Rider L" first="Lisa G." last="Rider">Lisa G. Rider</name><affiliation><nlm:aff id="A4">Lisa G. Rider, MD, NIEHS, NIH, Bethesda, MD, USA</nlm:aff></affiliation></author><author><name sortKey="Pilkington, Clarissa" sort="Pilkington, Clarissa" uniqKey="Pilkington C" first="Clarissa" last="Pilkington">Clarissa Pilkington</name><affiliation><nlm:aff id="A5">Clarissa Pilkington, MD, Great Ormond Street Hospital for Sick Children, London, United Kingdom</nlm:aff></affiliation></author><author><name sortKey="Oliveira, Sheila" sort="Oliveira, Sheila" uniqKey="Oliveira S" first="Sheila" last="Oliveira">Sheila Oliveira</name><affiliation><nlm:aff id="A6">Sheila Oliveira, MD, Prof, Universidade Federal do Rio de Janeiro, Instituto de Puericultura e Pediatria Martagao Gesteira, Rio de Janeiro, Brazil</nlm:aff></affiliation></author><author><name sortKey="Wulffraat, Nico" sort="Wulffraat, Nico" uniqKey="Wulffraat N" first="Nico" last="Wulffraat">Nico Wulffraat</name><affiliation><nlm:aff id="A7">Nico Wulffraat, MD, Wilhelmina Kinderziekenhuis, Utrecht, the Netherlands</nlm:aff></affiliation></author><author><name sortKey="Espada, Graciela" sort="Espada, Graciela" uniqKey="Espada G" first="Graciela" last="Espada">Graciela Espada</name><affiliation><nlm:aff id="A8">Graciela Espada, MD, Hospital de Ninos Ricardo Gutierrez, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Garay, Stella" sort="Garay, Stella" uniqKey="Garay S" first="Stella" last="Garay">Stella Garay</name><affiliation><nlm:aff id="A9">Stella Garay, MD, Hospital Sor Maria Ludovica, La Plata, Argentina</nlm:aff></affiliation></author><author><name sortKey="Cuttica, Ruben" sort="Cuttica, Ruben" uniqKey="Cuttica R" first="Ruben" last="Cuttica">Ruben Cuttica</name><affiliation><nlm:aff id="A10">Ruben Cuttica, MD, Hospital General de Ninos Pedro de Elizalde, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Hofer, Michael" sort="Hofer, Michael" uniqKey="Hofer M" first="Michael" last="Hofer">Michael Hofer</name><affiliation><nlm:aff id="A11">Michael Hofer, MD, Centre Multisite Romand de Rhumatologie Pediatrique, Lausanne, Switzerland</nlm:aff></affiliation></author><author><name sortKey="Quartier, Pierre" sort="Quartier, Pierre" uniqKey="Quartier P" first="Pierre" last="Quartier">Pierre Quartier</name><affiliation><nlm:aff id="A12">Pierre Quartier, MD, Universite Paris-Descartes, Hopital Necker-Enfants Malades, Paris, France</nlm:aff></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><nlm:aff id="A13">Jose Melo-Gomes, MD, Instituto Portugues de Reumatologia, Lisbon, Portugal</nlm:aff></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><nlm:aff id="A14">Ann M. Reed, MD, Mayo Clinic School of Medicine and Mayo Foundation, Rochester, MN, USA</nlm:aff></affiliation></author><author><name sortKey="Wierzbowska, Malgorzata" sort="Wierzbowska, Malgorzata" uniqKey="Wierzbowska M" first="Malgorzata" last="Wierzbowska">Malgorzata Wierzbowska</name><affiliation><nlm:aff id="A15">Malgorzata Wierzbowska, MD, Institute of Rheumatology, Warsaw, Poland</nlm:aff></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><nlm:aff id="A16">Brian M. Feldman, MD, MSc, FRCPC, University of Toronto, The Hospital for Sick Children, Toronto, Canada</nlm:aff></affiliation></author><author><name sortKey="Harjacek, Miroslav" sort="Harjacek, Miroslav" uniqKey="Harjacek M" first="Miroslav" last="Harjacek">Miroslav Harjacek</name><affiliation><nlm:aff id="A17">Miroslav Harjacek, MD, Children's Hospital Zagreb, Zagreb, Croatia</nlm:aff></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><nlm:aff id="A18">Hans-Iko Huppertz, MD, Klinikum Bremen-Mitte, Bremen, Germany</nlm:aff></affiliation></author><author><name sortKey="Nielsen, Susan" sort="Nielsen, Susan" uniqKey="Nielsen S" first="Susan" last="Nielsen">Susan Nielsen</name><affiliation><nlm:aff id="A19">Susan Nielsen, MD, Juliane Marie Centret, Rigshospitalet, Copenhagen, Denmark</nlm:aff></affiliation></author><author><name sortKey="Flato, Berit" sort="Flato, Berit" uniqKey="Flato B" first="Berit" last="Flato">Berit Flato</name><affiliation><nlm:aff id="A20">Berit Flato, MD, Rikshospitalet University Hospital, Oslo, Norway</nlm:aff></affiliation></author><author><name sortKey="Lahdenne, Pekka" sort="Lahdenne, Pekka" uniqKey="Lahdenne P" first="Pekka" last="Lahdenne">Pekka Lahdenne</name><affiliation><nlm:aff id="A21">Pekka Lahdenne, MD, Hospital for Children and Adolescents, Helsinki, Finland</nlm:aff></affiliation></author><author><name sortKey="Michels, Harmut" sort="Michels, Harmut" uniqKey="Michels H" first="Harmut" last="Michels">Harmut Michels</name><affiliation><nlm:aff id="A22">Harmut Michels, MD, Rheumakinderklinik Garmisch-Partenkirchen, Germany</nlm:aff></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><nlm:aff id="A23">Kevin J. Murray, MD, Princess Margaret Hospital for Children, Perth, WA, Australia</nlm:aff></affiliation></author><author><name sortKey="Punaro, Lynn" sort="Punaro, Lynn" uniqKey="Punaro L" first="Lynn" last="Punaro">Lynn Punaro</name><affiliation><nlm:aff id="A24">Lynn Punaro, MD, Texas Scottish Rite Hospital, Dallas, TX, USA</nlm:aff></affiliation></author><author><name sortKey="Rennebohm, Robert" sort="Rennebohm, Robert" uniqKey="Rennebohm R" first="Robert" last="Rennebohm">Robert Rennebohm</name><affiliation><nlm:aff id="A25">Robert Rennebohm, MD, Alberta Children's Hospital, Calgary, Canada</nlm:aff></affiliation></author><author><name sortKey="Russo, Ricardo" sort="Russo, Ricardo" uniqKey="Russo R" first="Ricardo" last="Russo">Ricardo Russo</name><affiliation><nlm:aff id="A26">Ricardo Russo, MD, Hospital de Pediatria Juan P. Garrahan, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Balogh, Zsolt" sort="Balogh, Zsolt" uniqKey="Balogh Z" first="Zsolt" last="Balogh">Zsolt Balogh</name><affiliation><nlm:aff id="A27">Zsolt Balogh, MD, National Institute of Rheumatology and Physiotherapy, Budapest, Argentina</nlm:aff></affiliation></author><author><name sortKey="Rooney, Madeleine" sort="Rooney, Madeleine" uniqKey="Rooney M" first="Madeleine" last="Rooney">Madeleine Rooney</name><affiliation><nlm:aff id="A28">Madeleine Rooney, MD, Musgrave Park Hospital, Belfast, Northern Ireland</nlm:aff></affiliation></author><author><name sortKey="Pachman, Lauren M" sort="Pachman, Lauren M" uniqKey="Pachman L" first="Lauren M." last="Pachman">Lauren M. Pachman</name><affiliation><nlm:aff id="A29">Lauren M. Pachman, MD, Children's Memorial Hospital, Chicago, IL, USA</nlm:aff></affiliation></author><author><name sortKey="Wallace, Carol" sort="Wallace, Carol" uniqKey="Wallace C" first="Carol" last="Wallace">Carol Wallace</name><affiliation><nlm:aff id="A30">Carol Wallace, MD, University of Washington/Children's Hospital, Seattle, WA, USA</nlm:aff></affiliation></author><author><name sortKey="Hashkes, Philip" sort="Hashkes, Philip" uniqKey="Hashkes P" first="Philip" last="Hashkes">Philip Hashkes</name><affiliation><nlm:aff id="A31">Philip Hashkes, MD, Shaare Zedek Medical Center, Jerusalem, Israel</nlm:aff></affiliation></author><author><name sortKey="Lovell, Daniel J" sort="Lovell, Daniel J" uniqKey="Lovell D" first="Daniel J." last="Lovell">Daniel J. Lovell</name><affiliation><nlm:aff id="A32">Daniel J. Lovell, MD, MPH, Edward H. Giannini, MSc, DrPH, Children's Hospital Medical Center, Cincinnati, OH, USA</nlm:aff></affiliation></author><author><name sortKey="Giannini, Edward H" sort="Giannini, Edward H" uniqKey="Giannini E" first="Edward H." last="Giannini">Edward H. Giannini</name><affiliation><nlm:aff id="A32">Daniel J. Lovell, MD, MPH, Edward H. Giannini, MSc, DrPH, Children's Hospital Medical Center, Cincinnati, OH, USA</nlm:aff></affiliation></author><author><name sortKey="Martini, Alberto" sort="Martini, Alberto" uniqKey="Martini A" first="Alberto" last="Martini">Alberto Martini</name><affiliation><nlm:aff id="A3">Angelo Ravelli, MD, Prof, Alberto Martini, MD, Prof, IRCCS G. Gaslini, Pediatria II, Reumatologia and Dipartimento di Pediatria, Università degli Studi, Genova, Italy</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">20583105</idno><idno type="pmc">2964396</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2964396</idno><idno type="RBID">PMC:2964396</idno><idno type="doi">10.1002/acr.20280</idno><date when="2010">2010</date><idno type="wicri:Area/Pmc/Corpus">001439</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">001439</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION 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><affiliation><nlm:aff id="A1">Nicolino Ruperto, MD, MPH, IRCCS G Gaslini, Pediatria II, Reumatologia, PRINTO, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Pistorio, Angela" sort="Pistorio, Angela" uniqKey="Pistorio A" first="Angela" last="Pistorio">Angela Pistorio</name><affiliation><nlm:aff id="A2">Angela Pistorio, MD, PhD, IRCCS G Gaslini, Servizio di Epidemiologia e Biostatistica, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Ravelli, Angelo" sort="Ravelli, Angelo" uniqKey="Ravelli A" first="Angelo" last="Ravelli">Angelo Ravelli</name><affiliation><nlm:aff id="A1">Nicolino Ruperto, MD, MPH, IRCCS G Gaslini, Pediatria II, Reumatologia, PRINTO, Genova, Italy</nlm:aff></affiliation><affiliation><nlm:aff id="A3">Angelo Ravelli, MD, Prof, Alberto Martini, MD, Prof, IRCCS G. Gaslini, Pediatria II, Reumatologia and Dipartimento di Pediatria, Università degli Studi, Genova, Italy</nlm:aff></affiliation></author><author><name sortKey="Rider, Lisa G" sort="Rider, Lisa G" uniqKey="Rider L" first="Lisa G." last="Rider">Lisa G. Rider</name><affiliation><nlm:aff id="A4">Lisa G. Rider, MD, NIEHS, NIH, Bethesda, MD, USA</nlm:aff></affiliation></author><author><name sortKey="Pilkington, Clarissa" sort="Pilkington, Clarissa" uniqKey="Pilkington C" first="Clarissa" last="Pilkington">Clarissa Pilkington</name><affiliation><nlm:aff id="A5">Clarissa Pilkington, MD, Great Ormond Street Hospital for Sick Children, London, United Kingdom</nlm:aff></affiliation></author><author><name sortKey="Oliveira, Sheila" sort="Oliveira, Sheila" uniqKey="Oliveira S" first="Sheila" last="Oliveira">Sheila Oliveira</name><affiliation><nlm:aff id="A6">Sheila Oliveira, MD, Prof, Universidade Federal do Rio de Janeiro, Instituto de Puericultura e Pediatria Martagao Gesteira, Rio de Janeiro, Brazil</nlm:aff></affiliation></author><author><name sortKey="Wulffraat, Nico" sort="Wulffraat, Nico" uniqKey="Wulffraat N" first="Nico" last="Wulffraat">Nico Wulffraat</name><affiliation><nlm:aff id="A7">Nico Wulffraat, MD, Wilhelmina Kinderziekenhuis, Utrecht, the Netherlands</nlm:aff></affiliation></author><author><name sortKey="Espada, Graciela" sort="Espada, Graciela" uniqKey="Espada G" first="Graciela" last="Espada">Graciela Espada</name><affiliation><nlm:aff id="A8">Graciela Espada, MD, Hospital de Ninos Ricardo Gutierrez, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Garay, Stella" sort="Garay, Stella" uniqKey="Garay S" first="Stella" last="Garay">Stella Garay</name><affiliation><nlm:aff id="A9">Stella Garay, MD, Hospital Sor Maria Ludovica, La Plata, Argentina</nlm:aff></affiliation></author><author><name sortKey="Cuttica, Ruben" sort="Cuttica, Ruben" uniqKey="Cuttica R" first="Ruben" last="Cuttica">Ruben Cuttica</name><affiliation><nlm:aff id="A10">Ruben Cuttica, MD, Hospital General de Ninos Pedro de Elizalde, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Hofer, Michael" sort="Hofer, Michael" uniqKey="Hofer M" first="Michael" last="Hofer">Michael Hofer</name><affiliation><nlm:aff id="A11">Michael Hofer, MD, Centre Multisite Romand de Rhumatologie Pediatrique, Lausanne, Switzerland</nlm:aff></affiliation></author><author><name sortKey="Quartier, Pierre" sort="Quartier, Pierre" uniqKey="Quartier P" first="Pierre" last="Quartier">Pierre Quartier</name><affiliation><nlm:aff id="A12">Pierre Quartier, MD, Universite Paris-Descartes, Hopital Necker-Enfants Malades, Paris, France</nlm:aff></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><nlm:aff id="A13">Jose Melo-Gomes, MD, Instituto Portugues de Reumatologia, Lisbon, Portugal</nlm:aff></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><nlm:aff id="A14">Ann M. Reed, MD, Mayo Clinic School of Medicine and Mayo Foundation, Rochester, MN, USA</nlm:aff></affiliation></author><author><name sortKey="Wierzbowska, Malgorzata" sort="Wierzbowska, Malgorzata" uniqKey="Wierzbowska M" first="Malgorzata" last="Wierzbowska">Malgorzata Wierzbowska</name><affiliation><nlm:aff id="A15">Malgorzata Wierzbowska, MD, Institute of Rheumatology, Warsaw, Poland</nlm:aff></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><nlm:aff id="A16">Brian M. Feldman, MD, MSc, FRCPC, University of Toronto, The Hospital for Sick Children, Toronto, Canada</nlm:aff></affiliation></author><author><name sortKey="Harjacek, Miroslav" sort="Harjacek, Miroslav" uniqKey="Harjacek M" first="Miroslav" last="Harjacek">Miroslav Harjacek</name><affiliation><nlm:aff id="A17">Miroslav Harjacek, MD, Children's Hospital Zagreb, Zagreb, Croatia</nlm:aff></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><nlm:aff id="A18">Hans-Iko Huppertz, MD, Klinikum Bremen-Mitte, Bremen, Germany</nlm:aff></affiliation></author><author><name sortKey="Nielsen, Susan" sort="Nielsen, Susan" uniqKey="Nielsen S" first="Susan" last="Nielsen">Susan Nielsen</name><affiliation><nlm:aff id="A19">Susan Nielsen, MD, Juliane Marie Centret, Rigshospitalet, Copenhagen, Denmark</nlm:aff></affiliation></author><author><name sortKey="Flato, Berit" sort="Flato, Berit" uniqKey="Flato B" first="Berit" last="Flato">Berit Flato</name><affiliation><nlm:aff id="A20">Berit Flato, MD, Rikshospitalet University Hospital, Oslo, Norway</nlm:aff></affiliation></author><author><name sortKey="Lahdenne, Pekka" sort="Lahdenne, Pekka" uniqKey="Lahdenne P" first="Pekka" last="Lahdenne">Pekka Lahdenne</name><affiliation><nlm:aff id="A21">Pekka Lahdenne, MD, Hospital for Children and Adolescents, Helsinki, Finland</nlm:aff></affiliation></author><author><name sortKey="Michels, Harmut" sort="Michels, Harmut" uniqKey="Michels H" first="Harmut" last="Michels">Harmut Michels</name><affiliation><nlm:aff id="A22">Harmut Michels, MD, Rheumakinderklinik Garmisch-Partenkirchen, Germany</nlm:aff></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><nlm:aff id="A23">Kevin J. Murray, MD, Princess Margaret Hospital for Children, Perth, WA, Australia</nlm:aff></affiliation></author><author><name sortKey="Punaro, Lynn" sort="Punaro, Lynn" uniqKey="Punaro L" first="Lynn" last="Punaro">Lynn Punaro</name><affiliation><nlm:aff id="A24">Lynn Punaro, MD, Texas Scottish Rite Hospital, Dallas, TX, USA</nlm:aff></affiliation></author><author><name sortKey="Rennebohm, Robert" sort="Rennebohm, Robert" uniqKey="Rennebohm R" first="Robert" last="Rennebohm">Robert Rennebohm</name><affiliation><nlm:aff id="A25">Robert Rennebohm, MD, Alberta Children's Hospital, Calgary, Canada</nlm:aff></affiliation></author><author><name sortKey="Russo, Ricardo" sort="Russo, Ricardo" uniqKey="Russo R" first="Ricardo" last="Russo">Ricardo Russo</name><affiliation><nlm:aff id="A26">Ricardo Russo, MD, Hospital de Pediatria Juan P. Garrahan, Buenos Aires, Argentina</nlm:aff></affiliation></author><author><name sortKey="Balogh, Zsolt" sort="Balogh, Zsolt" uniqKey="Balogh Z" first="Zsolt" last="Balogh">Zsolt Balogh</name><affiliation><nlm:aff id="A27">Zsolt Balogh, MD, National Institute of Rheumatology and Physiotherapy, Budapest, Argentina</nlm:aff></affiliation></author><author><name sortKey="Rooney, Madeleine" sort="Rooney, Madeleine" uniqKey="Rooney M" first="Madeleine" last="Rooney">Madeleine Rooney</name><affiliation><nlm:aff id="A28">Madeleine Rooney, MD, Musgrave Park Hospital, Belfast, Northern Ireland</nlm:aff></affiliation></author><author><name sortKey="Pachman, Lauren M" sort="Pachman, Lauren M" uniqKey="Pachman L" first="Lauren M." last="Pachman">Lauren M. Pachman</name><affiliation><nlm:aff id="A29">Lauren M. Pachman, MD, Children's Memorial Hospital, Chicago, IL, USA</nlm:aff></affiliation></author><author><name sortKey="Wallace, Carol" sort="Wallace, Carol" uniqKey="Wallace C" first="Carol" last="Wallace">Carol Wallace</name><affiliation><nlm:aff id="A30">Carol Wallace, MD, University of Washington/Children's Hospital, Seattle, WA, USA</nlm:aff></affiliation></author><author><name sortKey="Hashkes, Philip" sort="Hashkes, Philip" uniqKey="Hashkes P" first="Philip" last="Hashkes">Philip Hashkes</name><affiliation><nlm:aff id="A31">Philip Hashkes, MD, Shaare Zedek Medical Center, Jerusalem, Israel</nlm:aff></affiliation></author><author><name sortKey="Lovell, Daniel J" sort="Lovell, Daniel J" uniqKey="Lovell D" first="Daniel J." last="Lovell">Daniel J. Lovell</name><affiliation><nlm:aff id="A32">Daniel J. Lovell, MD, MPH, Edward H. Giannini, MSc, DrPH, Children's Hospital Medical Center, Cincinnati, OH, USA</nlm:aff></affiliation></author><author><name sortKey="Giannini, Edward H" sort="Giannini, Edward H" uniqKey="Giannini E" first="Edward H." last="Giannini">Edward H. Giannini</name><affiliation><nlm:aff id="A32">Daniel J. Lovell, MD, MPH, Edward H. Giannini, MSc, DrPH, Children's Hospital Medical Center, Cincinnati, OH, USA</nlm:aff></affiliation></author><author><name sortKey="Martini, Alberto" sort="Martini, Alberto" uniqKey="Martini A" first="Alberto" last="Martini">Alberto Martini</name><affiliation><nlm:aff id="A3">Angelo Ravelli, MD, Prof, Alberto Martini, MD, Prof, IRCCS G. Gaslini, Pediatria II, Reumatologia and Dipartimento di Pediatria, Università degli Studi, Genova, Italy</nlm:aff></affiliation></author></analytic><series><title level="j">Arthritis care & research</title><idno type="ISSN">2151-464X</idno><idno type="eISSN">2151-4658</idno><imprint><date when="2010">2010</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec id="S1"><title>Objective</title><p id="P1">To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (JDM) based on the PRINTO JDM core set of variables.</p></sec><sec sec-type="methods" id="S2"><title>Methods</title><p id="P2">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 (patients rating, statistical analysis, definition selection). Using the physicians’ consensus ratings as the “gold-standard measure”, chi-square, sensitivity, specificity, false positive and negative rate, area under the ROC, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa >0.8 were multiplied with the face validity score to select the top definitions.</p></sec><sec id="S3"><title>Results</title><p id="P3">The top definition of improvement was: at least 20% improvement from baseline in 3/6 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 3/6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength which is definition P1 selected by the IMACS group. The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process.</p></sec><sec id="S4"><title>Conclusion</title><p id="P4">we proposes 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 endpoint for the evaluation of global response to therapy in JDM.</p></sec></div></front></TEI><pmc article-type="research-article" xml:lang="EN"><pmc-comment>The publisher of this article does not allow downloading of the full text in XML form.</pmc-comment>
<pmc-dir>properties manuscript</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-journal-id">101518086</journal-id><journal-id journal-id-type="pubmed-jr-id">37048</journal-id><journal-id journal-id-type="nlm-ta">Arthritis Care Res (Hoboken)</journal-id><journal-title>Arthritis care & research</journal-title><issn pub-type="ppub">2151-464X</issn><issn pub-type="epub">2151-4658</issn></journal-meta><article-meta><article-id pub-id-type="pmid">20583105</article-id><article-id pub-id-type="pmc">2964396</article-id><article-id pub-id-type="doi">10.1002/acr.20280</article-id><article-id pub-id-type="manuscript">NIHMS218310</article-id><article-categories><subj-group subj-group-type="heading"><subject>Article</subject></subj-group></article-categories><title-group><article-title>THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION PROVISIONAL CRITERIA FOR THE EVALUATION OF RESPONSE TO THERAPY IN JUVENILE DERMATOMYOSITIS</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Ruperto</surname><given-names>Nicolino</given-names></name><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Pistorio</surname><given-names>Angela</given-names></name><xref ref-type="aff" rid="A2">2</xref></contrib><contrib contrib-type="author"><name><surname>Ravelli</surname><given-names>Angelo</given-names></name><xref ref-type="aff" rid="A1">1</xref><xref ref-type="aff" rid="A3">3</xref></contrib><contrib contrib-type="author"><name><surname>Rider</surname><given-names>Lisa G.</given-names></name><xref ref-type="aff" rid="A4">4</xref></contrib><contrib contrib-type="author"><name><surname>Pilkington</surname><given-names>Clarissa</given-names></name><xref ref-type="aff" rid="A5">5</xref></contrib><contrib contrib-type="author"><name><surname>Oliveira</surname><given-names>Sheila</given-names></name><xref ref-type="aff" rid="A6">6</xref></contrib><contrib contrib-type="author"><name><surname>Wulffraat</surname><given-names>Nico</given-names></name><xref ref-type="aff" rid="A7">7</xref></contrib><contrib contrib-type="author"><name><surname>Espada</surname><given-names>Graciela</given-names></name><xref ref-type="aff" rid="A8">8</xref></contrib><contrib contrib-type="author"><name><surname>Garay</surname><given-names>Stella</given-names></name><xref ref-type="aff" rid="A9">9</xref></contrib><contrib contrib-type="author"><name><surname>Cuttica</surname><given-names>Ruben</given-names></name><xref ref-type="aff" rid="A10">10</xref></contrib><contrib contrib-type="author"><name><surname>Hofer</surname><given-names>Michael</given-names></name><xref ref-type="aff" rid="A11">11</xref></contrib><contrib contrib-type="author"><name><surname>Quartier</surname><given-names>Pierre</given-names></name><xref ref-type="aff" rid="A12">12</xref></contrib><contrib contrib-type="author"><name><surname>Melo-Gomes</surname><given-names>Jose</given-names></name><xref ref-type="aff" rid="A13">13</xref></contrib><contrib contrib-type="author"><name><surname>Reed</surname><given-names>Ann M.</given-names></name><xref ref-type="aff" rid="A14">14</xref></contrib><contrib contrib-type="author"><name><surname>Wierzbowska</surname><given-names>Malgorzata</given-names></name><xref ref-type="aff" rid="A15">15</xref></contrib><contrib contrib-type="author"><name><surname>Feldman</surname><given-names>Brian M.</given-names></name><xref ref-type="aff" rid="A16">16</xref></contrib><contrib contrib-type="author"><name><surname>Harjacek</surname><given-names>Miroslav</given-names></name><xref ref-type="aff" rid="A17">17</xref></contrib><contrib contrib-type="author"><name><surname>Huppertz</surname><given-names>Hans-Iko</given-names></name><xref ref-type="aff" rid="A18">18</xref></contrib><contrib contrib-type="author"><name><surname>Nielsen</surname><given-names>Susan</given-names></name><xref ref-type="aff" rid="A19">19</xref></contrib><contrib contrib-type="author"><name><surname>Flato</surname><given-names>Berit</given-names></name><xref ref-type="aff" rid="A20">20</xref></contrib><contrib contrib-type="author"><name><surname>Lahdenne</surname><given-names>Pekka</given-names></name><xref ref-type="aff" rid="A21">21</xref></contrib><contrib contrib-type="author"><name><surname>Michels</surname><given-names>Harmut</given-names></name><xref ref-type="aff" rid="A22">22</xref></contrib><contrib contrib-type="author"><name><surname>Murray</surname><given-names>Kevin J.</given-names></name><xref ref-type="aff" rid="A23">23</xref></contrib><contrib contrib-type="author"><name><surname>Punaro</surname><given-names>Lynn</given-names></name><xref ref-type="aff" rid="A24">24</xref></contrib><contrib contrib-type="author"><name><surname>Rennebohm</surname><given-names>Robert</given-names></name><xref ref-type="aff" rid="A25">25</xref></contrib><contrib contrib-type="author"><name><surname>Russo</surname><given-names>Ricardo</given-names></name><xref ref-type="aff" rid="A26">26</xref></contrib><contrib contrib-type="author"><name><surname>Balogh</surname><given-names>Zsolt</given-names></name><xref ref-type="aff" rid="A27">27</xref></contrib><contrib contrib-type="author"><name><surname>Rooney</surname><given-names>Madeleine</given-names></name><xref ref-type="aff" rid="A28">28</xref></contrib><contrib contrib-type="author"><name><surname>Pachman</surname><given-names>Lauren M.</given-names></name><xref ref-type="aff" rid="A29">29</xref></contrib><contrib contrib-type="author"><name><surname>Wallace</surname><given-names>Carol</given-names></name><xref ref-type="aff" rid="A30">30</xref></contrib><contrib contrib-type="author"><name><surname>Hashkes</surname><given-names>Philip</given-names></name><xref ref-type="aff" rid="A31">31</xref></contrib><contrib contrib-type="author"><name><surname>Lovell</surname><given-names>Daniel J.</given-names></name><xref ref-type="aff" rid="A32">32</xref></contrib><contrib contrib-type="author"><name><surname>Giannini</surname><given-names>Edward H.</given-names></name><xref ref-type="aff" rid="A32">32</xref></contrib><contrib contrib-type="author"><name><surname>Martini</surname><given-names>Alberto</given-names></name><xref ref-type="aff" rid="A3">3</xref><on-behalf-of>for the Pædiatric Rheumatology International Trials Organisation (PRINTO) and the Pediatric Rheumatology Collaborative Study Group (PRCSG)</on-behalf-of></contrib></contrib-group><aff id="A1"><label>1</label>Nicolino Ruperto, MD, MPH, IRCCS G Gaslini, Pediatria II, Reumatologia, PRINTO, Genova, Italy</aff><aff id="A2"><label>2</label>Angela Pistorio, MD, PhD, IRCCS G Gaslini, Servizio di Epidemiologia e Biostatistica, Genova, Italy</aff><aff id="A3"><label>3</label>Angelo Ravelli, MD, Prof, Alberto Martini, MD, Prof, IRCCS G. Gaslini, Pediatria II, Reumatologia and Dipartimento di Pediatria, Università degli Studi, Genova, Italy</aff><aff id="A4"><label>4</label>Lisa G. Rider, MD, NIEHS, NIH, Bethesda, MD, USA</aff><aff id="A5"><label>5</label>Clarissa Pilkington, MD, Great Ormond Street Hospital for Sick Children, London, United Kingdom</aff><aff id="A6"><label>6</label>Sheila Oliveira, MD, Prof, Universidade Federal do Rio de Janeiro, Instituto de Puericultura e Pediatria Martagao Gesteira, Rio de Janeiro, Brazil</aff><aff id="A7"><label>7</label>Nico Wulffraat, MD, Wilhelmina Kinderziekenhuis, Utrecht, the Netherlands</aff><aff id="A8"><label>8</label>Graciela Espada, MD, Hospital de Ninos Ricardo Gutierrez, Buenos Aires, Argentina</aff><aff id="A9"><label>9</label>Stella Garay, MD, Hospital Sor Maria Ludovica, La Plata, Argentina</aff><aff id="A10"><label>10</label>Ruben Cuttica, MD, Hospital General de Ninos Pedro de Elizalde, Buenos Aires, Argentina</aff><aff id="A11"><label>11</label>Michael Hofer, MD, Centre Multisite Romand de Rhumatologie Pediatrique, Lausanne, Switzerland</aff><aff id="A12"><label>12</label>Pierre Quartier, MD, Universite Paris-Descartes, Hopital Necker-Enfants Malades, Paris, France</aff><aff id="A13"><label>13</label>Jose Melo-Gomes, MD, Instituto Portugues de Reumatologia, Lisbon, Portugal</aff><aff id="A14"><label>14</label>Ann M. Reed, MD, Mayo Clinic School of Medicine and Mayo Foundation, Rochester, MN, USA</aff><aff id="A15"><label>15</label>Malgorzata Wierzbowska, MD, Institute of Rheumatology, Warsaw, Poland</aff><aff id="A16"><label>16</label>Brian M. Feldman, MD, MSc, FRCPC, University of Toronto, The Hospital for Sick Children, Toronto, Canada</aff><aff id="A17"><label>17</label>Miroslav Harjacek, MD, Children's Hospital Zagreb, Zagreb, Croatia</aff><aff id="A18"><label>18</label>Hans-Iko Huppertz, MD, Klinikum Bremen-Mitte, Bremen, Germany</aff><aff id="A19"><label>19</label>Susan Nielsen, MD, Juliane Marie Centret, Rigshospitalet, Copenhagen, Denmark</aff><aff id="A20"><label>20</label>Berit Flato, MD, Rikshospitalet University Hospital, Oslo, Norway</aff><aff id="A21"><label>21</label>Pekka Lahdenne, MD, Hospital for Children and Adolescents, Helsinki, Finland</aff><aff id="A22"><label>22</label>Harmut Michels, MD, Rheumakinderklinik Garmisch-Partenkirchen, Germany</aff><aff id="A23"><label>23</label>Kevin J. Murray, MD, Princess Margaret Hospital for Children, Perth, WA, Australia</aff><aff id="A24"><label>24</label>Lynn Punaro, MD, Texas Scottish Rite Hospital, Dallas, TX, USA</aff><aff id="A25"><label>25</label>Robert Rennebohm, MD, Alberta Children's Hospital, Calgary, Canada</aff><aff id="A26"><label>26</label>Ricardo Russo, MD, Hospital de Pediatria Juan P. Garrahan, Buenos Aires, Argentina</aff><aff id="A27"><label>27</label>Zsolt Balogh, MD, National Institute of Rheumatology and Physiotherapy, Budapest, Argentina</aff><aff id="A28"><label>28</label>Madeleine Rooney, MD, Musgrave Park Hospital, Belfast, Northern Ireland</aff><aff id="A29"><label>29</label>Lauren M. Pachman, MD, Children's Memorial Hospital, Chicago, IL, USA</aff><aff id="A30"><label>30</label>Carol Wallace, MD, University of Washington/Children's Hospital, Seattle, WA, USA</aff><aff id="A31"><label>31</label>Philip Hashkes, MD, Shaare Zedek Medical Center, Jerusalem, Israel</aff><aff id="A32"><label>32</label>Daniel J. Lovell, MD, MPH, Edward H. Giannini, MSc, DrPH, Children's Hospital Medical Center, Cincinnati, OH, USA</aff><author-notes><corresp id="cor1">Please address correspondence and reprint requests to: Nicolino Ruperto, M.D., M.P.H., Pædiatric Rheumatology International Trials Organisation (PRINTO), IRCCS G. Gaslini, Università di Genova, Pediatria II - Reumatologia, EULAR Centre of Excellence in Rheumatology 2008-2013, Largo Gaslini, 5, 16147 Genova ITALY, Tel: +39-010-38-28-54 or +39-010-39-34-25, Fax: +39-010-39-33-24 or +39-010-39-36-19, <email>nicolaruperto@ospedale-gaslini.ge.it</email> <ext-link ext-link-type="uri" xlink:href="http://www.printo.it">http://www.printo.it</ext-link> or <ext-link ext-link-type="uri" xlink:href="http://www.pediatric-rheumatology.printo.it">www.pediatric-rheumatology.printo.it</ext-link></corresp></author-notes><pub-date pub-type="nihms-submitted"><day>16</day><month>7</month><year>2010</year></pub-date><pub-date pub-type="epub"><day>25</day><month>6</month><year>2010</year></pub-date><pub-date pub-type="ppub"><month>11</month><year>2010</year></pub-date><pub-date pub-type="pmc-release"><day>1</day><month>11</month><year>2011</year></pub-date><volume>62</volume><issue>11</issue><fpage>1533</fpage><lpage>1541</lpage><abstract><sec id="S1"><title>Objective</title><p id="P1">To develop a provisional definition for the evaluation of response to therapy in juvenile dermatomyositis (JDM) based on the PRINTO JDM core set of variables.</p></sec><sec sec-type="methods" id="S2"><title>Methods</title><p id="P2">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 (patients rating, statistical analysis, definition selection). Using the physicians’ consensus ratings as the “gold-standard measure”, chi-square, sensitivity, specificity, false positive and negative rate, area under the ROC, and kappa agreement for candidate definitions of improvement were calculated. Definitions with kappa >0.8 were multiplied with the face validity score to select the top definitions.</p></sec><sec id="S3"><title>Results</title><p id="P3">The top definition of improvement was: at least 20% improvement from baseline in 3/6 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 3/6 core set variables with no more than 2 of the remaining worsening by more than 25%, which cannot be muscle strength which is definition P1 selected by the IMACS group. The third is similar to the second with the maximum amount of worsening set to 30%. This indicates convergent validity of the process.</p></sec><sec id="S4"><title>Conclusion</title><p id="P4">we proposes 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 endpoint for the evaluation of global response to therapy in JDM.</p></sec></abstract><kwd-group><kwd>juvenile dermatomyositis</kwd><kwd>core set</kwd><kwd>response to therapy</kwd><kwd>disease activity</kwd><kwd>consensus</kwd></kwd-group><contract-num rid="ES1">Z01 ES101081-06
||ES</contract-num><contract-num rid="PHS2">NIH0010510991
||PHS</contract-num><contract-sponsor id="ES1">National Institute of Environmental Health Sciences : NIEHS</contract-sponsor><contract-sponsor id="PHS2">NIH Intramural Employee : PHS</contract-sponsor></article-meta></front></pmc></record>Pour manipuler ce document sous Unix (Dilib)
EXPLOR_STEP=$WICRI_ROOT/Wicri/Asie/explor/AustralieFrV1/Data/Pmc/Corpus
HfdSelect -h $EXPLOR_STEP/biblio.hfd -nk 001439 | SxmlIndent | more
Ou
HfdSelect -h $EXPLOR_AREA/Data/Pmc/Corpus/biblio.hfd -nk 001439 | SxmlIndent | more
Pour mettre un lien sur cette page dans le réseau Wicri
{{Explor lien
|wiki= Wicri/Asie
|area= AustralieFrV1
|flux= Pmc
|étape= Corpus
|type= RBID
|clé= PMC:2964396
|texte= THE PEDIATRIC RHEUMATOLOGY INTERNATIONAL TRIALS ORGANIZATION PROVISIONAL CRITERIA FOR THE EVALUATION OF RESPONSE TO THERAPY IN JUVENILE DERMATOMYOSITIS
}}
Pour générer des pages wiki
HfdIndexSelect -h $EXPLOR_AREA/Data/Pmc/Corpus/RBID.i -Sk "pubmed:20583105" \
| HfdSelect -Kh $EXPLOR_AREA/Data/Pmc/Corpus/biblio.hfd \
| NlmPubMed2Wicri -a AustralieFrV1
| This area was generated with Dilib version V0.6.33. |  |