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STUDYPROTOCOLOpenAccessDevelopmentofaninternationallyagreedminimaldatasetforjuveniledermatomyositis(JDM)forclinicalandresearchuseLizaJ.
McCann1*,JamieJ.
Kirkham2,LucyR.
Wedderburn3,4,5,ClarissaPilkington4,5,AdamM.
Huber6,AngeloRavelli7,DuncanAppelbe2,PaulaR.
Williamson2andMichaelW.
Beresford1,8AbstractBackground:Juveniledermatomyositis(JDM)isarareautoimmuneinflammatorydisorderassociatedwithsignificantmorbidityandmortality.
Internationalcollaborationisnecessarytobetterunderstandthepathogenesisofthedisease,responsetotreatmentandlong-termoutcome.
Toaidinternationalcollaboration,itisessentialtohaveacoresetofdatathatallresearchersandclinicianscollectinastandardisedwayforclinicalpurposesandforresearch.
Thisshouldincludedemographicdetails,diagnosticdataandmeasuresofdiseaseactivity,investigationsandtreatment.
VariablesinexistingclinicalregistrieshavebeencomparedtoproduceaprovisionaldatasetforJDM.
Wenowaimtodevelopthisintoaconsensus-approvedminimumcoredataset,testedinawidersetting,withtheobjectiveofachievinginternationalagreement.
Methods/Design:Atwo-stagebespokeDelphi-processwillengagetheopinionofalargenumberofkeystakeholdersthroughEmaildistributionviaestablishedinternationalpaediatricrheumatologyandmyositisorganisations.
This,togetherwithaformalisedpatient/parentparticipationprocesswillhelpinformaconsensusmeetingofinternationalexpertsthatwillutiliseanominalgrouptechnique(NGT).
Theresultingproposedminimaldatasetwillbetestedforfeasibilitywithinexistingdatabaseinfrastructures.
Thedevelopedminimaldatasetwillbesenttoallinternationallyrepresentativecollaboratorsforfinalcomment.
Theparticipantsoftheexpertconsensusgroupwillbeaskedtodrawtogetherthesecomments,ratifyand'signoff'thefinalminimaldataset.
Discussion:Aninternationallyagreedminimaldatasethasthepotentialtosignificantlyenhancecollaboration,alloweffectivecommunicationbetweengroups,provideaminimalstandardofcareandenableanalysisofthelargestpossiblenumberofJDMpatientstoprovideagreaterunderstandingofthisdisease.
Thefinalapprovedminimumcoredatasetcouldberapidlyincorporatedintonationalandinternationalcollaborativeefforts,includingexistingprospectivedatabases,andbeavailableforuseinrandomisedcontrolledtrialsandfortreatment/protocolcomparisonsincohortstudies.
Keywords:Juvenile,Childhood,Dermatomyositis,Idiopathicinflammatorymyopathy,International,Collaboration,Dataset,Coreoutcomeset,Coredataset,Diseaseregister,Clinicaltrials,Diseaseactivity*Correspondence:liza.
mccann@alderhey.
nhs.
uk1AlderHeyChildren'sNHSFoundationTrust,EatonRoad,Liverpool,UKFulllistofauthorinformationisavailableattheendofthearticleTRIALS2015McCannetal.
ThisisanOpenAccessarticledistributedunderthetermsoftheCreativeCommonsAttributionLicense(http://creativecommons.
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0),whichpermitsunrestricteduse,distribution,andreproductioninanymedium,providedtheoriginalworkisproperlycredited.
TheCreativeCommonsPublicDomainDedicationwaiver(http://creativecommons.
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McCannetal.
Trials(2015)16:268DOI10.
1186/s13063-015-0784-0BackgroundItisrecognisedthataninternationallyagreedcoredata-setforuseinroutineclinicalcarecouldhavenumerouspotentialbenefitsforpatientswiththisraredisease,withtheadditionaladvantageofinformingfutureresearch[1].
Thegoalsofthisstudyaretoreachinternationalconsensusonaminimaldataset(clinical,laboratoryandpatient/parentreportedmeasures)forpatientswithju-veniledermatomyositis(JDM),whicharepractical,butcoverthekeyvariablesthatallowaccurateassessmentofdiseaseactivityandwouldmeasurechange,suchasre-sponsetotreatment.
Suchacoresetcouldthenberapidlyincorporatedintonationalandinternationalcollaborativeefforts.
Todate,2distinctbutoverlappingcoresetsforJDMhavebeendevelopedforuseinclinicaltrialsbytheInternationalMyositisandClinicalStudies(IMACS)group[2–4]andthePaediatricRheumatologyInter-nationalTrialsOrganisation(PRINTO)[5–7].
Theseex-cellenttoolshavebeenprincipallydesignedforresearchandcanbedifficulttouseinroutineclinicalpracticeduetotimeconstraints.
Ouraimistocomplimentthesecoresetswithaseparateinternationallyagreedminimaldatasetforuseinclinicalpractice.
Thekeyachievableobjectiveofthisstudyisforthedevelopedminimaldatasettoberecognisedandacceptedinternationallytoallowdatacol-lectionovertimeonsufficientnumbersofJDMpatients.
Inordertoachievethis,aformalconsensus-drivenmeth-odologyisrequiredofallstakeholders.
Backgroundworktothisproposalisdescribedelsewhere[1].
Inbrief,aworkinggroupoffiveJDMexperts(CP,LW,AH,AR,LM)fromtheUK,ItalyandCanada,repre-sentativeofthemajorgroupsstudyingJDMandmaintain-ingdatabases,scrutinisedclinicalandlaboratoryvariablescontainedwithincurrentnationalandinternationalcol-laborativedatabases.
Variablescontainedwithin2ormoredatabaseswereconsideredforinclusionintheprovisionalminimaldataset,informedbyaliteraturesearchandde-tailedanalysisoftheUKJuvenileDermatomyositisCohortandBiomarkerStudy(JDCBS)[1,8].
DetailsofthisprojecthavebeenincludedintheCoreOutcomeMeasuresinEffectivenessTrials(COMET)ini-tiativedatabase[9,10].
InadditiontoclosecollaborationwithCOMET,ourgrouphassoughtadvicefromseniormembersoftheOMERACT(OutcomeMeasuresinRheumatology)collaboration,whoarerenownedfortheirworkrelatingtooutcomestandardisation[11,12].
Methods/DesignAprovisionaldatasetforJDM,developedthroughafor-malprocess[1]hasbeenusedasatemplatetoaidastructuredmulti-stageconsensusprocess.
TheDelphitechnique[13–15]hasbeenusedtogaintheopinionoflargegroupsofinternationallyrepresentativeclinicianscaringforchildrenandyoungpeoplewithJDM.
This,togetherwithastepwiseapproachtogaintheopinionofpatients/parents,willhelpinformadetailedface-to-facenominalgroupconsensusprocessbyinternationallyrep-resentativeJDMexperts.
Stage1a–DelphiprocesstogainclinicianopinionA2-stagesurveytechnique(Delphi1andDelphi2;witheachstagebuildingontheproceedingone)hasbeenadopted,sothatanonymousresponsesareobtainedwithequalinfluencetoallindividualparticipants[13–15].
Aninitiallistofallpotentialvariableswasobtainedfromtheprovisionalminimaldatabaseproducedbythisgroup[1]withoutcomeslistedindividuallybutgroupedundertherelevantdomaintoaidinterpretationbyclinicians.
Out-comesidentifiedaspatient/parentreportedoutcomemeasuresorvalidatedtoolsofmusclestrengthmeasure-ment[16]werepresentedwithintheappropriatedomainasuseofthetoolratherthanasindividualcomponentsofthetool.
However,inordertoavoiddiscussionsatthisstagearoundwhichtool/instrumentisbetterformeas-urementasopposedtoanothertool,aninitialquestionaskedparticipantshowimportanttheyratetheuseofatool(withoutspecifyingwhichtool)tomeasureacertaindomain,suchasmusclestrengthorskininvolvement.
Iftheyconsideredatooltobeimportant,theywereaskedwhichtooltheyfavoured.
Thelistofvariableswasreviewedandapprovedbyallauthorsaswellastheresearchsteeringcommitteeorleadingrepresentativesofeachoftheprincipalpartnerorganisations(detailedinTable1),includingtheInter-nationalMyositisandClinicalStudiesgroup–IMACS[4],ChildhoodArthritisandRheumatologyResearchAl-liance–CARRA[17],JuvenileDermatomyositisRe-searchGroup–JDRG(UKandIreland),[18],PaediatricRheumatologyEuropeanSociety–PReSJDMworkingparty[19],andthePaediatricRheumatologyINter-nationalTrialsOrganisation–PRINTO[7].
Aftermodi-ficationsaddressingfeedbackfromthesegroups,thelist(showninTable2)wasformattedintoabespokeelec-tronicquestionnaire.
TheUKPaediatricRheumatologyTrainees'GroupandindividualsfromoutsidetheUKse-lectedbymembersoftheJDMMinimalDatasetSteeringCommittee,pilotedthequestionnairetodetermineaveragetimetakentocompleteandreadabilityofthequestions.
ParticipantdetailsParticipationfortheDelphisurveyswasinvitedviamembershipofIMACS,CARRA,JDRG,PReSJDMworkingpartyandPRINTO;representativeofinter-nationalpaediatricrheumatologyandmyositisspecialtygroups.
Theseorganisationsincludeclinicians(clinicalacademicandnon-academic),scientistsandalliedhealthprofessionalswithexpertiseinpaediatricrheumatologicalconditionsandjuvenile-onsetandadult-onsetmyositisMcCannetal.
Trials(2015)16:268Page2of15Table1DetailsofcollaborativegroupsandpartnerorganisationsCollaborativeorganisationMembershipMaingeographicalareacoveredCollaborativeroleinstudyJuvenileDermatomyositisResearchGroup,JDRG(UKandIreland)[18]Multi-disciplinaryhealthcareprofessionalsandscientistsinthefieldofPaediatricRheumatology;representativeofallmajorUKpaediatricrheumatologycentres.
JDRGmembersareallcontributorsto,andinvestigatorsin,theJuvenileDermatomyositisCohortandBiomarkerStudy,JDCBS[8]UKandIrelandDelphisurveyEmaildistributionRepresentationinNGTmeetingScrutinisingdatacollection(Stage3)AiddisseminationofstudyresultsSteeringcommittee–studyoversightPatient/parentrepresentation:steeringcommitteeandfacilitatedgroupsChildhoodArthritisandRheumatologyResearchAlliance,CARRA[17]PaediatricrheumatologistsandresearchersthroughoutNorthAmerica(n>390);aproportionofwhomhaveaninterestinJDMandothersmoreexperiencedinotherareasofpaediatricrheumatologyNorthAmericaDelphisurveysEmaildistributionRepresentationinNGTmeetingandprovisionalminimaldatasetAiddisseminationofstudyresultsPaediatricRheumatologyINternationalTrialsOrganisation,PRINTO[7]InternationalresearchnetworkwiththeaimofcoordinatingclinicaltrialsandoutcomestudiesinpaediatricrheumatologyInitiallyaEuropeancollaborationbutnowincludes>50countriesand>350centresworldwideDelphisurveysviaE-maildistributionRepresentationinNGTmeetingandprovisionalminimaldatasetAiddisseminationofstudyresultsPaediatricRheumatologyEuropeanSociety,PReS[19]Europeanscientificsocietyforhealthcareprofessionalsinthefieldofpaediatricrheumatology.
ThePReSJDMworkinggroupisasub-groupofthisorganisationthatinvitesindividualswithaninterestorexpertiseinJDMAllEuropeancountries(EUandnon-EU),extendedtotheMiddleEast.
AssociatememberswelcomeworldwideDelphisurveysviaEmaildistributionRepresentationinNGTmeetingAiddisseminationofstudyresultsInternationalMyositisAssessmentandClinicalStudiesgroup,IMACS[4]Coalitionofhealthcareprovidersandresearcherswithaninterestinmyositissyndromes.
MyositissyndromesincludeJDMbutalsootherinflammatorymyopathiesthataremorecommoninadultpatientsPartoftheNIHscienceresearchprogramme,USAbutwelcomesmembersgloballyDelphisurveysviaEmaildistributionRepresentationinNGTmeetingAiddisseminationofstudyresultsEuromyositis[39,40]Europeaninitiativeleadingtothecreationofaweb-basedregistryforadultmyositispatients,recentlyexpandedtoincludepaediatricsInitiallyEuropeancounties(EUandnon-EU)expandedtoincludeothercollaboratorsoutsideEurope(Japan,Mexico,China,US)CollaboratorsinformationofaminimaldatasetRepresentationinNGTmeetingTestingdatacollectionovertime(Stage3,potential)OMERACT[11]AnindependentinitiativeofinternationalhealthprofessionalsinterestedinoutcomemeasuresinrheumatologyNorthAmerica,EuropeandAsia-PacificAdvisoryroleindevelopmentofDelphisurveyandinpatientinvolvementRepresentationinNGTmeetingJDM,juveniledermatomyositis;NGT,nominalgrouptechnique;NIH,NationalInstituteforHealthMcCannetal.
Trials(2015)16:268Page3of15Table2SummaryofquestionsaskedincliniciansurveycomparedtotheparentandpatientquestionnairesDomainQuestionsaskedofhealthcareprofessionalsinDelphi1and2QuestionsaskedinparentquestionnaireQuestionsaskedinyoungperson'squestionnaireGeneralformatofquestionnaireTwoseparatesections:SectionA–whenpatientseenfortheirfirstvisitonly;SectionB–allvisits(firstandsubsequent).
Askedtorateimportanceofeachvariableona1–9scaleseparatelyforclinicaluseandforresearchAskedtorate:HowimportantdoyouthinkeachoneoftheseiswhenthinkingabouthowwellorunwellyourchildisduetoJDM("notthatimportant"/"important"/or"reallyimportant").
Notaskedseparatelyaboutclinical/researchAskedtorate:HowimportantdoyouthinkeachoneoftheseiswhenthinkingabouthowwellorunwellyouareduetoJDM("notthatimportant"/"important"/"reallyimportant")Notaskedseparatelyaboutclinical/researchGeneralquestionsoncollectingandstoringinformationand/orspecimensN/AAskedtoratehowimportanttheythinkitisforalldoctorsandnurses/therapiststocollectkeyinformationinthesamewaywhentheyarelookingafterchildrenwithjuveniledermatomyositis(JDM)Askedtoratehowimportanttheythinkitisforalldoctorsandnurses/therapiststocollectkeyinformationinthesamewaywhentheyarelookingafterchildrenwithJDMHowimportantdoyouratestoringspecimensatdiagnosisforotherbiomarkers(eg.
,DNA/serum/anyothermaterial)aAskedtoratehowimportanttheythinkitistostorethetypeofinformationcollectedinclinicinaresearchdatabaseAskedtoratehowimportanttheythinkitistostorethetypeofinformationcollectedinclinicinaresearchdatabaseDemographicsofpatientDateofbirthN/AN/AGenderRaceEthnicityFamilyhistoryOfautoimmunediseaseN/AN/AOfneuromusculardiseaseDiagnosticdata(general)AgeofonsetofJDMN/AN/AAgeofdiagnosisofJDMMUSCLE–Diagnostic/activitydataPresenceofsymmetricalproximalmuscleweakness:atdiagnosis(sectionA)andeveryvisit(sectionB)AskingyouifyourchildhasanyweaknessAskingifyouhaveanyweaknessPresenceofothermuscleweakness(eg.
,distalorasymmetrical):sectionBonlyUseofavalidatedtooltoscoremusclestrengthand/orfunction(optionsfordifferentmusclestrengthscores)aTestingtoseehowstrongyourchildisTestingtoseehowstrongyouareSKIN–Diagnostic/activitydataHeliotroperash(sectionsAandB)AskingyouifyourchildhasanyskinrashesAskingifyouhaveanyskinrashesGottron'spapules/Gottron'ssign(sectionsAandB)Nail-foldcapillarychanges(sectionsAandB)OthercharacteristicJDMrashUseofavalidatedskintoolforJDM(optionsgiventochoosespecifictools)aLookingforrashesorskinsignsthatmaysuggestactiveJDMLookingforrashesorskinsignsthatmaysuggestactiveJDMSKIN–AdditionalactivitydataaskedinsectionBonlyLipodystrophyN/AN/ACalcinosisCutaneousulcerationMcCannetal.
Trials(2015)16:268Page4of15Table2Summaryofquestionsaskedincliniciansurveycomparedtotheparentandpatientquestionnaires(Continued)SubcutaneousoedemaMalar/facialerythemaShawlsign/V-signMechanic'shandsAlopeciaVasculopathiclesionsPhotosensitivityLividoreticularisOthererythemaPanniculitisMAJORORGANINVOLVEMENTduetomyositis–askedinsectionBonlyofcliniciansurvey,pluspatient/parentquestionnairesMusculoskeletalinvolvement(specificquestionsaddedforarthritis/contractures)aAskingifyourchildhasanymuscleorjointpainsAskingifyouhaveanymuscleorjointpainsLookingforswellinginanyofthejointsLookingforswellinginanyofthejointsGastrointestinalinvolvement(specificquestionsaddedfordysphagia/abdominalpainorGIulceration)aAskingifyourchildhasanydifficultywithswallowing/eatingoriftheyhavetummypainAskingifyouhaveanydifficultywitheatingorifyouhavetummypainPulmonaryinvolvementsuggestinginterstitiallungdiseaseAskingifyourchildhasanyshortnessofbreathorchestpainthatmayberelatedtoJDMAskingifyouhaveanyshortnessofbreathorchestpainthatmayberelatedtoJDMCardiacinvolvementCONSTITUTIONALSYMPTOMSduetomyositis–askedinsectionBonlyofcliniciansurveyandinpatient/parentquestionnaireFever(>38°C)duetomyositisN/AN/AWeightloss(>5%)duetomyositisN/AN/AFatigueduetomyositisAskingifyourchildfeelstiredduetoJDMAskingifyoufeeltiredduetoJDMAskinghowtiredyourchildfeelsusingaformal"fatiguescale"Askinghowtiredyoufeelusingaformal'fatiguescale'IrritabilityduetomyositisAskingifyourchildfeelsirritableormiserableduetoJDMAskingifyoufeelirritableormiserableduetoJDMRaynaud'sphenomenonAskingifyourchildgetscolourchangesintheirhandsincoldweatherAskingifyougetcolourchangesinyourhandsincoldweatherGrowthHeightofpatient(sectionBonly)Checkinghowwellyourchildisgrowing(height)CheckinghowwellyouaregrowingWeightofpatient(sectionBonly)Askingaboutanyweightlossandcheckingyourchild'sweightAskingaboutanyweightlossandcheckingyourweightDevelopment/pubertyN/AAskingabouthowyourchildisdevelopingorhowpubertyisprogressingAskingabouthowyouaredevelopingorhowpubertyisprogressingOtherinformationPresenceofmalignancy(sectionBonly)N/AN/APatienthavingongoingfollow-upatthiscentre(Ifno:optionsgivenforreasonwhy)aGlobaldiseaseactivityAskingyourdoctortomarkona0–10cmscalehowwellorunwelltheythinkyourchildhasbeenAskingyourdoctortomarkona0–10cmscalehowwellorunwelltheythinkyouhavebeenMcCannetal.
Trials(2015)16:268Page5of15Table2Summaryofquestionsaskedincliniciansurveycomparedtotheparentandpatientquestionnaires(Continued)Useofaphysician-scoredmeasureofglobaldiseaseactivity-suchasphysicianVisualAnalogueScale;VAS(specificchoicesforactivityscoresgiven)abasedonwhatyoutellthemandwhattheyseeatyourclinicvisitPatient/parentReportedOutcomeMeasures(PROMs),QualityofLife/SchoolissuesUseofapatient/parentreportedoutcomemeasureofdiseaseactivityoruseofavalidatedmeasureoffunction(suchaspatientVAS/CHAQ)and/oratooltomeasureQualityofLife(QoL)(specificchoicesgiven)aAskingyouoryourchildtocompleteaquestionnairethatlooksathoweasyordifficultitisforthemtodothingslikegetdressed,haveabath,doactivitiesAskingyouortocompletealistofquestionsthatlookathoweasyordifficultitisforyoutodothingslikegetdressed,haveabath,doactivitiesAskingyou/yourchildtomarkona0–10cmscalehowwellorunwellyourchildhasbeenoverthelast4weeksAskingyoutomarkona0–10cmscalehowwellorunwellyouhavebeenoverthelast4weeksduetoJDMAskingyou/yourchildtomarkona0–-10cmscalehowmuchpainyourchildhashadinthelast4weeksduetoJDMAskingyoutomarkona0–10cmscalehowmuchpainyouhavehadinthelast4weeksduetoJDMAskinghowmanydaysyourchildhasmissedschoolorcollegeduetoJDMAskinghowmanydaysyouhavemissedschoolorcollegeduetoJDMAskingmorequestionsaboutyourchild'sschool–howarethingsAretheyabletokeepupwithpeersAskingmorequestionsaboutschool–howarethingsAreyouabletokeepupwithyourpeersAskingyourchildhowtheyfeelemotionallyinrelationtotheirJDM(questionsrelatingtoQualityofLifeormood)AskingaboutyourfeelingsinrelationtoyourJDMINVESTIGATIONS:diagnosticdata/diseaseactivitydataElevationofmuscleenzymesatdiagnosis(sectionA)orlaterindiseasecourse(sectionB).
(Questionsaskedaboutwhichspecificenzymestomeasure)aTakingbloodteststomonitorhowactivethediseaseisTakingbloodteststomonitorhowactivethediseaseisElectromyography(EMG)changesofmyositisatdiagnosis(sectionA)Musclebiopsyevidenceofmyositisatdiagnosis(sectionA)MagneticResonanceImaging(MRI)changesofmyositisatdiagnosis(sectionA)AskingyourchildtohaveascansuchasanMRIscanoftheirmusclestomonitorthediseaseAskingyoutohaveascansuchasanMRIscanofyourmusclesSectionBonly:abnormalinvestigations(imaging,histologyorcardio-pulmonaryfunctiontestsindicatingflareofmyositis).
If"yes,"tickwhichinvestigationsareabnormalAnti-nuclearAntibody(ANA)positivityatdiagnosis(sectionAonly)Myositisspecificantibody(MSA)positiveatdiagnosis(sectionAonly)Myositisassociatedantibody(MAA)positivityatdiagnosis(sectionAonly)TreatmentDatetreatmentstarted(mm/yyyy)Askingspecificquestionsaboutyourchild'smedicinesandhowtheymakethemfeelAnyunwantedeffectsAskingspecificquestionsaboutyourmedicinesandhowtheymakeyoufeelAnyunwantedeffectsMcCannetal.
Trials(2015)16:268Page6of15Table2Summaryofquestionsaskedincliniciansurveycomparedtotheparentandpatientquestionnaires(Continued)PatienttakingsteroidsYes/No(optionsfortypeofsteroidgiven)aAskingyouaboutthemedicinesyourchildistakingatthemomentAskingyouaboutthemedicinesyouaretakingatthemomentPatienttakingaDiseaseModifyingAnti-RheumaticDrug(DMARD)(NotincludingbiologicDMARDS)Yes/No(withoptiontoselectnameofdrugfromalist)PatienttakingabiologicYes/No(withoptiontoselectnameofdrugfromalist)Patienthavingphysiotherapyand/oroccupationaltherapyAskingyouifyourchildisdoinganyphysiotherapyexercisesAskingyouifyouaredoingyourphysiotherapyexercisesOtherquestionsTodeterminepracticeandexperience-primaryrole,patientgroup,numberofpaediatric/adultpatientsundertheircare,geographicalregionofpracticeandmembershipofsocieties/professionalbodiesHowwouldyoupreferinformationtobeaskedofyou(eg.
,inclinic,questionnairebeforeclinic,questionnairebeforeyoucometoclinic)Howwouldyoupreferinformationtobeaskedofyou(eg.
,inclinic,questionnairebeforeclinic,questionnairebeforeyoucometoclinic)FurtherinformationArethereanyvariablesthatyouthinkareimportantforclinicalcarethatarenotcurrentlyincludedinthelistaboveIfso,pleasestatewhattheseare(samequestionalsoaskedseparatelyforvariablesimportantforresearch)Arethereanyotherkeyquestionsthatwehavemissedbutthatyouthinkitisimportantforustoaskwhenyoucometoclinic,thinkingabouthowwell/unwellyou/yourchildisIfso,whatkeyquestionsaretheyArethereanyotherkeyquestionsthatwehavemissedbutthatyouthinkareimportantforustoaskwhenyoucometoclinic,thinkingabouthowwellorunwellyouareIfso,whatkeyquestionsaretheyIsthereanythinginthislist(orfromyourexperienceofbringingyourchildtoclinic)thatyoudonotthinkshouldbeincludedoryouthinkyourchilddoesnotlikeIfso,whatWhydoyouthinkthatthesequestionsshouldnotbeincludedIsthereanythinginthislist(orfromyourexperienceofcomingtoclinic)thatyoudonotlikeIfso,whatWhydoyouthinkthatthesequestionsshouldnotbeincludedaFurtherdetailedquestionsaskedinhiddenrows,exposediftheanswertotheprecedingquestionsuggeststhattheparticipantthinksthatthevariableisimportantN/A,notapplicableMcCannetal.
Trials(2015)16:268Page7of15(Table1).
TheentiremembershipwascontactedbyEmailandinvitedtoparticipateinthetwoweb-basedquestion-nairesusingabespokeDelphisystem.
TheonlyexceptiontothiswasthePRINTOmembership.
InviewofthefactthatPRINTOisalargeandheterogeneousorganisationwithover900individualmembers,thePRINTOdirectors(approximately400members)werecontacted.
PRINTOdirectorsrepresentallofthemajorpaediatricrheumatol-ogycentresamongmembercountries.
ManyEmailrecipi-entsarepartofmorethanoneresearchorganisationandinthiscase,theywereaskedtocompleteeachsurveyroundonceonlyandidentifywhichothergroupstheyaremembersof(and,therefore,potentiallypartofmorethanonemailing).
Delphiround1Thestudyobjectiveswereclearlystatedincludingwhatwasexpectedofeachparticipant.
ParticipantswereremindedoftheimportanceofcompletingtheentireDel-phiprocessandaskedtocompleteeachroundwithin6weeksofreceiptofEmail.
ReminderEmailsweresentonafortnightlybasistoaidcompletionofeachround,withphrasingofremindersconsideredtomaximisethere-sponserates[20].
Participantswhoagreedtotakepartwereaskedtoregister,allowingauniqueidentifiertobeallocatedtoenabletrackingofattritionateachroundandidentificationoftheresearchgroupthattheparticipantwasrespondingfrom(IMACS,CARRA,PRINTO,PReSJDMworkinggrouportheUKJDRG).
Contributorswereaskedtospecifytheirpredominantclinicalrole(clinician/scientist/alliedhealthprofessional/trainee),theirspecialty(specialistormajorinterestinrheumatology/neurology/dermatology/immunology/other),theageofpatientthattheypredominantlycarefor(adult/paediatric)theirexperi-enceinthespecialty/condition(≥or16yearsofage)whohaveorpreviouslyhadjuvenile-onsetmyositisandparentsofchildrenwithJDM.
NHSResearchEthicsapprovalobtainedStage2(nominalgroupconsensus)doesnotinvolvepatientdataandsimplyinvolvesconsensusamongexpertsinmyositisStage3willinvolveanalysisofanonymiseddataafterpilotingtheminimaldatasetinaproportionofpatientsoverarestrictedperiodoftime(6months)viaalreadyMcCannetal.
Trials(2015)16:268Page12of15establisheddatabases.
Ethicalapprovalforthisstageiscountryspecific,andalthoughitwillinvolveanonymisedpatientdata,itisrequiredforsomecollaborators.
EthicalapprovalisalreadyobtainedfordatacollectionwithinmostoftheorganisationsthatmaycollaborateatthisstageincludingtheUKJDCBS,Euromyositis,theGasliniInstituteandCARRA.
OtherparticipantswillbeabletoenterdataviaEuromyositisorobtaintheirownIRBapproval.
However,thestudyisnotdependentonIRBapprovaloftheseindividualcountries,assufficientpatientnumbersshouldbeachievedviauseofestablisheddatabasesthatalreadyincludeethicalapproval.
Ourgroupwillonlyhaveaccess,throughresearchsteeringcommitteeapplication,tosecondaryanonymiseddatacollectedwithintheseindividualdatabases.
DiscussionTheDelphiprocesshastheadvantageofallowinginfor-mationtobeexchangedbetweennumerousindividualswhomaybegeographicallydispersed[38].
However,theadvantageofface-to-faceinteractionisthatitallowsidentificationofthereasonsfordisagreementsbetweenindividuals[38].
ByusingacombinationofDelphiweb-basedsurveysandface-to-faceinteractioninanNGTconsensusgroupprocess,thisstudywillobtainwide-spreadopinionfromalargenumberofcliniciansinadditiontothejudgmentofasmallgroupofinter-nationallyrenownedandrepresentativegroupofexpertsinJDM.
Patientandparentparticipationwillbeensuredviaastructuredprocess,includingwidespreaddistribu-tionofaquestionnaireandmoredetaileddiscussionwithinestablishedpatient/parentgroupsintheUK.
Theuseoffocusgroupswillallowstimulationofdiscussionandcomparisonofexperiencesacrossparticipants,whereasindividualquestionnaireresponseswillallowobtainmentofopinionswithoutinfluencefrompeers[30,41].
Inaddition,considerationwillbegiventoasmallnumberofpatient/parentrepresentativespartici-patingintheconsensusmeeting,representingtheviewsofthisgroup.
Aninternationallyagreedminimaldatasethasthepo-tentialtosignificantlyenhancecollaboration,allowef-fectivecommunicationbetweengroups,provideaminimalstandardofcareandenableanalysisofthelar-gestpossiblenumberofJDMpatientstoprovideagreaterunderstandingofthisdisease.
Aconsensus-driven,internationallyapprovedminimumcoredatasetcouldberapidlyincorporatedintonationalandinter-nationalcollaborativeefforts,includingexistingpro-spectiveclinicaldatabases,andbeavailableforuseinrandomisedcontrolledtrialsandfortreatment/protocolcomparisonsincohortstudies.
StudystatusCurrentlyatStage3:Delphisurveyofcliniciansandconsensusmeetingcompleted.
Patient/parentquestion-nairesongoing.
AbbreviationsCRN:ClinicalResearchNetwork;CSG:ClinicalStudiesGroup;BSPAR:BritishSocietyforPaediatricandAdolescentRheumatology;CARRA:ChildhoodArthritisandRheumatologyResearchAlliance;COMET:CoreOutcomeMeasuresinEffectivenessTrialsinitiative;GCP:GoodClinicalPractice;GRADE:GradeofRecommendationsAssessment,DevelopmentandEvaluation;JDCBS:JuvenileDermatomyositisCohortBiomarkerStudyandRepository(UKandIreland);JDM:juveniledermatomyositis;JDRG:JuvenileDermatomyositisResearchGroup(UKandIreland);IMACS:InternationalMyositisandClinicalStudiesgroup;MCRN:MedicinesforChildrenResearchNetwork;NIHR:NationalInstituteforHealthResearch;NGT:nominalgrouptechnique;OMERACT:OutcomeMeasuresinRheumatology;PReS:PaediatricRheumatologyEuropeanSociety;PRINTO:PaediatricRheumatologyINternationalTrialsOrganisation;SHARE:SingleHubandAccessPointforPaediatricRheumatologyinEurope;TSG:TopicSpecificGroup.
CompetinginterestsTheauthorsdeclarethattheyhavenocompetinginterests.
Authors'contributionAllauthorshavebeendirectlyinvolvedindevelopingthemethodologyforthisstudy.
LMhasledallpartsofthestudyincludingbackgroundwork,preparationoftheprotocol,ethicssubmissions,contentofDelphisurveyandpatient/parentquestionnaires,planningofconsensusmeetingandwritingofthemanuscript.
JKparticipatedinthedesignofthestudy,wasresponsiblefortestingtheDelphisystem,performedthestatisticalanalysisandhelpedpreparefortheconsensusmeeting.
LW,CP,AHandARhaveprovidedintellectualinputandpracticalhelpintoallpartsofthestudyincludingbackgroundwork,protocoldevelopment,Delphisurveyandplanningoftheconsensusmeeting.
DAdevelopedthebespokeDelphisystemandprovidedITsupportforthestudyincludingdataanalysis.
PWhasprovidedexpertadviceonthestudymethodologyandanalysis.
MBhasbeenresponsibleforintellectualandfinancialoverviewofthestudyandinputintotheprotocoldevelopment,Delphisurveyandconsensusmeeting.
Allauthorshavereadandapprovedthefinalmanuscript.
Authors'informationDrLizaMcCannisaconsultantinpaediatricrheumatologyatAlderHeyChildren'sNHSFoundationTrust,UK,witharesearchinterestinjuveniledermatomyositis.
DrJamieKirkhamisalecturerinbiostatisticsattheUniversityofLiverpool,InstituteofTranslationalMedicine,UK.
ProfessorLucyWedderburnisProfessorofPaediatricRheumatologyattheInstituteofChildHealth,UniversityCollegeLondon;consultantatGreatOrmondStreetHospital;ChiefInvestigatoroftheJuvenileDermatomyositisCohortandBiomarkerStudy,(JDCBS)UKandIrelandandDirectorofArthritisResearchUKCentreforAdolescentRheumatologyatUCL.
DrClarissaPilkingtonisaconsultantinpaediatricandadolescentrheumatologyatGreatOrmondStreetandUniversityCollegeHospital,London,UK;PresidentoftheBritishSocietyforPaediatricandAdolescentRheumatology.
Shehasaspecialinterestinjuveniledermatomyositisandsystemiclupuserythematosis.
DrAdamHuberisapaediatricrheumatologistattheIWKHealthCentreandAssociateProfessoratDalhousieUniversity,withaparticularresearchinterestinjuvenilemyositis.
ProfessorAngeloRavelliisAssociateProfessorofPaediatricsattheUniversityofGenoaandIstitutoGianninaGaslini,Genoa,Italy.
Hisresearchinterestsincludejuvenilemyositis.
DrDuncanAppelbeistheInformationSystemsManagerattheMCRNClinicalTrialsUnit,UniversityofLiverpoolInstituteofTranslationalMedicine,UK.
ProfessorPaulaWilliamsonisHeadofDepartmentandDirectoroftheClinicalTrialsResearchCentreattheUniversityofLiverpoolInstituteofTranslationalMedicine.
McCannetal.
Trials(2015)16:268Page13of15ProfessorMichaelWBeresfordisBroughChairandProfessorofChildHealth,UniversityofLiverpool;AcademicLeadintheClinicalAcademicDepartmentofPaediatricRheumatologyatAlderHeyChildren'sNHSFoundationTrust,LiverpoolUKandChairoftheUK'sNationalInstituteofHealthResearchClinicalResearchNetwork:Children/ArthritisResearchUKPaediatricRheumatologyClinicalStudiesGroup.
AcknowledgmentsThisworkwassupportedbyArthritisResearchUK[grantnumber20417].
WewouldliketoacknowledgetheEuromyositisResearchSteeringCommitteefortheircollaborationandimpetusforthiswork,inparticularProfessorIngridLundberg,DrHectorChinoy,ProfessorJiriVencovskyandNielsSteenKrogh.
WeacknowledgetheIMACS,CARRA,PRINTOandPReSworkinggroupsfortheirsupportandcollaboration.
Inparticular,wewouldliketoacknowledgethefollowingcollaboratorswhorepresentthesegroups:DrLisaRider,ProfessorCharlesSpencer,DrNicolaRupertoandDrAnnetvanRoyen-Kerkhof.
WeacknowledgethesupportofOMERACTandinparticular,wouldliketothankProfessorMaartenBoersforhisadvisoryrole.
WewouldliketoacknowledgeourcollaboratorswithintheCOMETgroup,inparticular,HeatherBagley,PatientandPublicInvolvementCoordinator.
WewouldalsoliketoacknowledgethehelpandadviceofProfessorBridgetYoung,OliviaLloydandHelenHansonandtheClinicalStudiesGroupconsumerrepresentativesandBSPARParentGroup,particularlySharonDouglas.
WewouldliketothankyoungpeoplefromNIHRYoungPerson'sAdvisoryGroupandtheJDMYoungPerson'sGroup,fortheiradviceonpatientquestionnairesandinformationleaflets.
Weacknowledgethesupportandcollaborationofpatient/parentsupportgroups,CureJMandMyositisUK.
WewouldliketothankKatieArnold,LawrenceBrown,KathForrestandKarenBarnesfortheiradministrativesupportforthiswork.
TheUKJDMCohortandBiomarkerstudyhasbeensupportedbygenerousgrantsfromtheWellcomeTrustUK(085860),ActionMedicalResearchUK,(SP4252),TheMyositisSupportGroupUK,ArthritisResearchUK(14518),TheHenrySmithCharity.
LWsworkissupportedinpartbyGreatOrmondStreetChildren'sCharity,theGOSH/ICHNIHRfundedBiomedicalresearchCentre(BRC)andArthritisResearchUK.
TheJDMCohortstudyisadoptedontotheComprehensiveResearchNetworkthroughtheMedicinesforChildrenResearchNetwork(www.
mcrn.
org.
uk)andissupportedbytheGOSH/ICHBiomedicalResearchCentre.
AfulllistofcontributorstotheJDCBScanbefoundontheJDRGwebsitehttp://www.
juveniledermatomyositis.
org.
uk.
Authordetails1AlderHeyChildren'sNHSFoundationTrust,EatonRoad,Liverpool,UK.
2MRCNorthWestHubforTrialsMethodologyResearch,DepartmentofBiostatistics,UniversityofLiverpool,Liverpool,UK.
3Infection,Immunology,andRheumatologySectionUCLInstituteofChildHealth,UniversityCollegeLondon,London,UK.
4GreatOrmondStreetHospitalNHSFoundationTrust,London,UK.
5CentreforAdolescentRheumatologyatUniversityCollegeLondon,UniversityCollegeLondonHospital,London,UK.
6IWKHealthCentreandDalhousieUniversity,5850UniversityAvenue,Halifax,NSB3K6R8,Canada.
7UniversitàdegliStudidiGenovaandIstitutoGianninaGaslini,ViaG.
Gaslini5,16147Genoa,Italy.
8DepartmentofWomen'sandChildren'sHealth,InstituteofTranslationalMedicine,UniversityofLiverpool,Liverpool,UK.
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