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The University of Manchester Research Increasing the Value of Orthodontic Research Through the Use of Dental Patient-Reported Outcomes DOI: 10.1016/j.jebdp.2019.04.005 Document Version Accepted author manuscript Link to publication record in Manchester Research Explorer Citation for published version (APA): Hua, F. (2019). Increasing the Value of Orthodontic Research Through the Use of Dental Patient-Reported Outcomes. Journal of Evidence-Based Dental Practice. https://doi.org/10.1016/j.jebdp.2019.04.005 Published in: Journal of Evidence-Based Dental Practice Citing this paper Please note that where the full-text provided on Manchester Research Explorer is the Author Accepted Manuscript or Proof version this may differ from the final Published version. If citing, it is advised that you check and use the publisher's definitive version. General rights Copyright and moral rights for the publications made accessible in the Research Explorer are retained by the authors and/or other copyright owners and it is a condition of accessing publications that users recognise and abide by the legal requirements associated with these rights. Takedown policy If you believe that this document breaches copyright please refer to the University of Manchester’s Takedown Procedures [http://man.ac.uk/04Y6Bo] or contact [email protected] providing relevant details, so we can investigate your claim. Download date:16. Oct. 2020

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Page 1: Increasing the Value of Orthodontic Research Through the ......Nowadays, patients’ psycho-social function is considered a core element of oral health. Also, their treatment needs

The University of Manchester Research

Increasing the Value of Orthodontic Research Through theUse of Dental Patient-Reported OutcomesDOI:10.1016/j.jebdp.2019.04.005

Document VersionAccepted author manuscript

Link to publication record in Manchester Research Explorer

Citation for published version (APA):Hua, F. (2019). Increasing the Value of Orthodontic Research Through the Use of Dental Patient-ReportedOutcomes. Journal of Evidence-Based Dental Practice. https://doi.org/10.1016/j.jebdp.2019.04.005

Published in:Journal of Evidence-Based Dental Practice

Citing this paperPlease note that where the full-text provided on Manchester Research Explorer is the Author Accepted Manuscriptor Proof version this may differ from the final Published version. If citing, it is advised that you check and use thepublisher's definitive version.

General rightsCopyright and moral rights for the publications made accessible in the Research Explorer are retained by theauthors and/or other copyright owners and it is a condition of accessing publications that users recognise andabide by the legal requirements associated with these rights.

Takedown policyIf you believe that this document breaches copyright please refer to the University of Manchester’s TakedownProcedures [http://man.ac.uk/04Y6Bo] or contact [email protected] providingrelevant details, so we can investigate your claim.

Download date:16. Oct. 2020

Page 2: Increasing the Value of Orthodontic Research Through the ......Nowadays, patients’ psycho-social function is considered a core element of oral health. Also, their treatment needs

Increasing the Value of Orthodontic Research Through the Use of Dental Patient-Reported Outcomes

FangHuaa,b

aCenterforEvidence-BasedStomatology&DepartmentofOrthodontics,Hubei-MOSTKLOS&

KLOBM,School&HospitalofStomatology,WuhanUniversity,Wuhan,ChinabDivisionofDentistry,SchoolofMedicalSciences,FacultyofBiology,MedicineandHealth,The

UniversityofManchester,ManchesterAcademicHealthScienceCentre,Manchester,UK

Correspondingauthor:

Fang Hua, School & Hospital of Stomatology, Wuhan University, 237 Luoyu Road, Wuhan

430079,China

E-mail:[email protected]

How to Cite:

Hua F. Increasing the Value of Orthodontic Research Through the Use of Dental

Patient-Reported Outcomes. Journal of Evidence-Based Dental Practice, 2019. doi:

10.1016/j.jebdp.2019.04.005 [Epub ahead of print].

doi: 10.1016/j.jebdp.2019.04.005

Please note that this is a post-print version (after peer-review) for the purpose of self-

archiving. Please download the final published version from the publisher’s website:

https://www.sciencedirect.com/science/article/pii/S1532338219300806

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Abstract

Tenyearsago,ChalmersandGlaszioupointedoutinLancetthat85%ofallbiomedical

researchwasbeingavoidablywastedduetoimbalancedresearchquestion/outcome

selection,aswellaspoorstudydesign,executionandreporting.Accordingtofindings

ofrecent“researchonresearch”studies,ahighlevelofsuchkindofavoidablewaste

shouldalsoexistinorthodonticresearch.Thiswarrantseffortstoimproveeachstage

oftheresearchproductionandreportingprocess.

Nowadays,patients’psycho-socialfunctionisconsideredacoreelementoforalhealth.

Also, their treatment needs and preferences have formed a main component of

evidence-baseddentistry(EBD).Therefore,inordertoachieveshareddecision-making,

orthodontistsneedtohaveanadequateunderstandingofeachpatient’svaluesand

perceptions.

In this context, orthodontic research should be patient-centered so that the

knowledgeandperceptiongapsbetweencliniciansandpatientscanbereduced,and

research evidence that is suitable for patients to understand can be provided. In

addition, patient-centered outcomes that can reflect patients’ perceptions and

psycho-socialstatusshouldbewidelyused.

However, recent scoping reviewshaveshownthatduring thepastdecade,patient-

centeredoutcomesincludingthoseregardingadverseeffects,healthserviceresource

utilizationandqualityof lifehaveremainedunderrepresented inorthodontictrials.

Thus, the use of dental patient-reported outcomes (dPROs) and dental patient-

reportedoutcomemeasures(dPROMs)shouldbepromotedtofacilitatetheprovision

ofapatient-centeredevidencebase,reducetheavoidablewasterelatedtoresearch

question/outcomeselection,andtherebyincreasethevalueoforthodonticresearch.

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AvoidableWasteinOrthodonticResearch

Justtenyearsago,ChalmersandGlasziou1pointedoutinLancetthatamajorityofall

biomedicalresearchwasbeingavoidablywastedduetoimbalancedresearchquestion

/outcomeselection,inadequatestudydesignandexecution,non-publication,lackof

accessibility,andpoorreporting.Asover50%ofstudiesfailedtotakeadequatesteps

toreducebiases(e.g.inadequateallocationconcealment),2over50%ofstudieswere

never published in full,3 andmore than50%of planned studyoutcomeswerenot

reported,4theyestimatedthattheproportionofsuchwastewasgreaterthan85%.1

Thereafter, the Lancet launched theREWARD (REduce researchWasteAndReward

Diligence)campaignandpublishedaseriesoffivereviewsregarding“increasingvalue

and reducing waste”, each covering one stage in the production and reporting of

biomedicalresearch(Figure).5,6

Inorthodontics,recent“researchonresearch”studieshavefoundthatabout70%of

randomizedcontrolledtrials(RCTs)didnothaveadequateallocationconcealment,750%

ofstudieswereneverfullypublished,8andmorethan80%ofRCTsdidnotadequately

reporttheirpre-specifiedprimaryoutcomemeasures.9Basedonthesefindingsand

thecalculationmethodusedbyChalmersandGlasziou,1theestimatedproportionof

avoidablewasteinorthodonticresearchwouldbeevenhigherthan85%.Thislooks

appalling,butstillithasnottakenintoaccountthefactthatover60%oforthodontic

RCTswerefocusedonoutcomesthatwereirrelevanttopatients,10andthatmorethan

30%oforthodonticpaperswerenotfreelyavailableonlineforcliniciansandpatients

(Figure).11

Undoubtedly,suchahighlevelofavoidablewastewarrantseffortstoimproveallfive

stagesoftheproductionandreportingoforthodonticresearch.Thisarticlewilldiscuss

potentialmeasurestotakeforthefirststageand,morespecifically,thesignificanceof

patient-centered research and dental patient-reported outcomes (dPROs) for

orthodontics.

Patients’PerceptionsandtheDefinitionofOralHealth

Recognition of the importance of patients’ perception can be traced back to the

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additionof“socialwell-being”intotheWHOdefinitionofhealthin1940s,whichled

to a paradigm shift in the model of health from biomedical to biopsychosocial.12

Thereafter,similarmodificationsweremadetothedefinitionoforalhealth.In1993,

Dolan definedoral health (for older populations) as “a comfortable and functional

dentitionwhich allows individuals to continue in their desired social role”.13More

recently,theFDIWorldDentalFederationdevelopedanewdefinitionfororalhealth:

“Oralhealthismultifacetedandincludestheabilitytospeak,smile,smell,taste,

touch,chew,swallow,andconveyarangeofemotionsthroughfacialexpressions

with confidence and without pain, discomfort, and disease of the craniofacial

complex.”14

Inaddition,theFDIWorldDentalFederationemphasizedthatoralhealth“reflectsthe

physiological,socialandpsychologicalattributesthatareessentialtothequalityoflife

(QoL),” and “is influenced by the person’s changing experiences, perceptions,

expectations, and ability to adapt to circumstances.”14 These definitions have all

importantlyindicatedthatthegoalsofdentalhealthcareshouldincludenotonlythe

treatmentofpatients’diseases/conditions,butalsotheimprovement/restoration

oftheirphysiologicalandpsycho-socialfunctions.Toachievethese,itisessentialthat

dentalprofessionalsincludingorthodontistshaveanadequateunderstandingoftheir

patients’perceptionsandexpectations,bothatanindividuallevelandapopulation

level.

Evidence-BasedMedicineandEvidence-BasedDentistry

A common criticismof evidence-basedmedicine (EBM)has been that EBMmeans

“cookbook medicine” and decision-making based on evidence alone, without

necessarydeliberationandreasoning.15However,asexpertsinEBM16andevidence-

based orthodontics (EBO) 17 have pointed out, such an argument is merely a

misunderstandingofwhatEBMis.Ineffect,theneedtoconsidereachpatient’svalues

hasbeenaggressivelypromotedsincetheveryearlyyearsofEBM.16

In1996,Sacketandcolleaguesalreadystatedthat:

“EBMistheconscientious,explicit,andjudicioususeofcurrentbestevidencein

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makingdecisionsaboutthecareofindividualpatients.ThepracticeofEBMmeans

integrating individual clinical expertisewith the best available external clinical

evidence from systematic research. … Increased expertise is reflected inmany

ways, but especially inmore effective and efficient diagnosis and in themore

thoughtful identification and compassionate use of individual patients’

predicaments, rights, and preferences in making clinical decisions about their

care.”18

Monthslater,theywentontoproposeasimplemodelforevidence-baseddecision-

making,withclinicians’expertise,patients’preferencesandresearchevidencebeing

thethreemaincomponentsofthismodel.19In1999,thesecomponentswereadopted

bytheAmericanDentalAssociation(ADA)foritsofficialdefinitionofevidence-based

dentistry(EBD):

“EBDisanapproachtooralhealthcarethatrequiresthejudiciousintegrationof

systematic assessments of clinically relevant scientific evidence, relating to the

patient’s oral and medical condition and history, with the dentist’s clinical

expertiseandthepatient’streatmentneedsandpreferences.”20,21

Theemphasis thatEBMandEBDplaceonpatients is inaccordancewithacultural

change inmedicine over the past decades: the increasing importance of patients’

autonomyandconsent,aswellastheassociateddemandforshareddecision-making

(SDM)andpatient-centeredcare.16,22

SharedDecision-MakinginOrthodonticPractice

AccordingtoGatellarietal,23SDMisadecision-makingprocessinwhichcliniciansand

patientsareequalpartnersandarerequiredtoexchangeinformationandpreferences

in order to negotiate a mutually acceptable treatment decision. In the case of

orthodontics,thisapproachisparticularlyimportantformanyreasons.Someofthe

mainonesareasfollows.

First, as malocclusion is not a disease but a variation from the societal norm,24

orthodontictreatmentisoftenelective.25Plus,orthodontictreatmentisnotwithout

risks / adverse effects (e.g. root resorption, enamel demineralization).26 The SDM

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approachcanfacilitatethoroughexchangeofexpectations,preferencesandpossible

treatment outcomes between orthodontists and patients, and thereby help them

determinewhetherorthodontictreatmentisabletoachievetheintendedeffect,and

whetherthepotentialgainoutweighrisksandcosts.

Second,orthodonticpatientsusuallyexpecttoimprovetheirdentofacialappearance

through orthodontic treatment.27 However, the patients’ perception of esthetics is

oftendifferent from thatof orthodontists. For instance, patients seem tobemore

tolerant / less sensitive than orthodontists towards gummy smile,28 midline

discrepancies,29andwhitespotlesions.30Inlightofsuchdiscrepancies,orthodontists

shouldusetheSDMapproachtoobtainanadequateunderstandingofthepatient’s

expectationandtreatmentneed.Apersonalizedtreatmentgoalthatisacceptableto

bothpartiescouldthenbesetandachieved.

Inaddition,likeinotherdentalspecialties,foreachorthodonticproblemthereusually

exist multiple therapies / techniques that can be considered.31 Examples include

differenttypesofappliances(e.g.conventionalbrackets,self-ligatingbrackets,clear

aligners),methodstorelievecrowding(e.g.toothextraction,interproximalreduction,

total arch distalization), and methods to reinforce anchorage (e.g. miniscrews,

transpalatalarch,headgear).However,sincemanysystematicreviewsinorthodontics

failedtoperformmeaningfulmeta-analyses(mostlyduetolowquantityandquality

of eligible studies, as well as the use of heterogeneous outcome measures),

orthodontistsareoftenfacedwithindefiniteorconflictingevidencewhencomparing

theavailable therapies/ techniques.32 In thiskindofsituations,SDMisparticularly

needed so that clinician’s expertise and patients’ values / preferences can bewell

integratedandleadtothemostsuitableclinicaldecision.33

Patient-CenteredResearchandPatient-CenteredOutcomes

Theultimategoalofallmedicalresearchistoimproveourknowledgeunderpinning

thepreventionandtreatmentofillness.34InthecontextofEBD,dentalstudiesshould

bedesigned,executedandreportedproperly,sothattheirfindingsarerelevant,valid

and usable, facilitating the achievement and development of patient-centered,

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evidence-baseddentalpractice.Towards thisend, the researchquestionsofdental

studiesshouldberelevanttopatients,theoutcomesthattheyuseshouldbeableto

reflectpatients’valuesandperceptions.35

In2013,TsichlakiandO’Brien10carriedoutascopingreviewtoanalyzetheoutcomes

usedinorthodonticRCTsduring2008-2012.Theyfoundthat63%ofthesetrialswere

focusedonassessingmorphologicchangesofmalocclusion;whereaspatient-centered

outcomesincludingthoseregardingadverseeffects,healthserviceresourceutilization

andQoLwereonlymeasuredin32%,32%and9%oftheincludedtrials,respectively.

In2017,Tsichlakiandcolleagues36updatedtheabove-mentionedscopingreview,as

the first stage of developing a core outcome set (i.e. an agreed, minimum set of

outcomestobemeasuredandreported)fororthodontictrials.37Atotalof54different

outcomescovering14outcomedomainswereidentified,withhalfoftheoutcomes

assessedbymultipleoutcomemeasures (range:2 to6).Althoughpatient-reported

painwasthemostfrequentlyusedoutcome(18%),theauthorsfoundthatmostofthe

outcomeswere still focusedon the assessment ofmorphologic changes,withQoL

measuredbyonly4(2%)oftheincludedtrials.

These data have shown a problem with the current selection of outcomes in

orthodontic studies.AspointedoutbyO’Brien,38 although those clinician-centered

outcomesthatorthodonticresearcherstraditionallymeasure(e.g.changesinoverjet

ortheANBangle)canbeeasilycollectedfromstudycasts,radiographs,patientrecords

andclinicalexaminations,theycanneitherreflectpatients’perceptionsandvalues,

nor demonstrate how well an intervention can improve the patient’s oral health.

Studiesthatonlyassessclinician-centeredoutcomesmaybeignoringormissingthe

acceptabilityofeachintervention,i.e.thebenefitsanddrawbacksofanintervention

fromthepatient’sperspective.

InanarticlepublishedinTheNewEnglandJournalofMedicine,Porter39proposeda

three-tiered hierarchy of outcome measures that can cover health circumstances

relevanttopatients,includingthehealthstatusachievedorretained(tier1),process

ofrecovery(tier2),andsustainabilityofhealth(tier3).Heemphasizedthatthevalue

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ofhealthcareshouldalwaysbedefinedaroundpatients,andtheonlywaytomeasure

this value accurately is to track patient outcomes and costs longitudinally, with a

follow-uplongenoughtoencompasstheultimateresultsofcare.

Furthermore,inordertoachieveandimproveSDM,weneedmorepatient-centered

researchthatcanreducethegapinknowledgeandperceptionsbetweencliniciansand

patients, providing research evidence that is suitable for patients to understand.31

PromotingtheuseofdPROsinorthodonticresearchcouldbeonestepfurthertowards

theprovisionofsuchapatient-centeredevidencebase.25

Dental Patient-Reported Outcomes and Dental Patient-ReportedOutcomeMeasures

dPROs,atermrecentlycoinedbyJohn,40aredefinedas“anyreportofthestatusofa

patient’s oral health condition that comes directly from the patient, without

interpretationof thepatient’s responsebyaclinicianoranyoneelse.” Just likeoral

healthisacomponentofgeneralhealth,dPROsareasubsetofallpatient-reported

outcomes(PROs).dPROscancapturetheimpactofconditionsorinterventionsonoral

health,whiledentalpatient-reportedoutcomemeasures(dPROMs)areusedtoassess

thisimpactandexpressitnumerically.40

Inarecentsystematicreview,Johnandcolleagues41analyzedtheexistingmulti-item,

genericdPROMsforadultdentalpatients.Basedon20eligiblequestionnaires(e.g.the

OralHealthImpactProfile),theyidentified53dPROMs,whichrepresentallcurrently

availablemulti-iteminstrumentsforassessingtheperceivedoralhealthofpatients.A

total of 36 unique dPROswere assessed by these dPROMs, including 35 narrower

dPROs (e.g. functional limitations, physical pain, psychological discomfort) and 1

broader dPRO – the oral health-related quality of life (OHRQoL). Also, the authors

found that those 35 narrower dPROs can be categorized into four groups: Oral

Function, Orofacial Appearance, Orofacial Pain, and Psychosocial Impact. This is in

accordancewiththefourdimensionsofOHRQoL,42,43indicatingthatthesedPROshave

coveredallmajorconcernsofdentalpatientswiththeiroralhealth.41

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In an earlier article of this series, Reissmann 44 introduced several additional

advantages of dPROMs over disease-oriented outcome measures for research: 1)

better reliability and the resultant smaller required sample size; 2) flexible

administrationandeasydatacollection;3) lowerworkloadandhigherefficiency;4)

providinginformationabouttheentirepatient;and5)easyinterpretationandsample

sizecalculation(whentheminimalimportantdifferenceisavailable).

OralHealth-RelatedQualityofLifeandOrthodonticResearch

Asmentionedabove,OHRQoLisamajordPRO,andeachOHRQoLinstrumentcanbe

seenasadPROMoracombinationofseveraldPROMs(dependingonthenumberof

dimensions the instrument contains).41 During the past twenty years, orthodontic

researchershavecarriedoutmanyepidemiologicalstudiesregardingOHRQoL.45Based

onthese,recentsystematicreviewshaveconcludedthatmalocclusionhasasignificant

(thoughsmall)negativeimpactontheOHRQoLofchildren(mainlyintheemotional

and socialwell-being dimensions),46-48 and that orthodontic treatment can lead to

moderate but significant improvement in OHRQoL.49, 50 Despite the low quality

evidence underlying these reviews (mainly due to inadequate study design and

substantial heterogeneity), they have demonstrated the capability of OHRQoL to

reflectorthodontictreatmentneedandthebenefitsoforthodonticinterventionsto

patients. Unfortunately, so far very few clinical studies in orthodontics have used

OHRQoL as their primary or secondary outcome, especially those RCTs.10, 36

Nevertheless,anopportunitytosolvethisproblemmayariseinthenearfuture,once

the core outcome set for orthodontic research (which includes OHRQoL related

dPROMs)iscompletedandavailable.37

Inaddition,anotherrelevantissueisthelargenumberofOHRQoLinstrumentsthat

havebeenusedinorthodonticresearch.Forinstance,amongstudieswithregardto

the association between malocclusion and OHRQoL, at least eight different

instrumentshavebeenused.47Suchalackofstandardizationbringsdifficultiestothe

interpretationandsynthesesofdifferentstudies,andisthereforeunhelpfulforSDM

and patient-centered care (even though dPROMs have been used). Again, a core

outcomesetmaybethekeytothisissueifitincludesonlyoneOHRQoLinstrument

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for each possible situation (e.g. different age groups). Such an instrumentmay be

selected /modified fromtheexisting toolsbasedon their scopeandpsychometric

properties,43, 44 or be developed specifically for the context of malocclusion /

orthodonticsbasedonperceptionsofthetargetpopulation.51,52

Summary

Patients’psycho-socialfunctionisacoreelementoforalhealth;theirtreatmentneeds

andpreferencesformamaincomponentofEBD.However,recentstudieshaveshown

thatmostoftheoutcomesmeasuredinorthodontictrialsarefocusedonassessing

morphologic changes, whereas patient-centered outcomes have remained

underrepresented.Inlightofthis,theuseofdPROsanddPROMsshouldbepromoted

tofacilitatetheprovisionofapatient-centeredevidencebase,reducetheavoidable

waste related to research question / outcome selection, and thereby increase the

valueoforthodonticresearch.

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Acknowledgements

FHissupportedbytheChinaPostdoctoralScienceFoundation(No.2018M630884).

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FigureLegend

Figure.ThefivestagesofresearchwasteproposedbyChalmersandcolleagues1,5,6

andrelevantfindingsfrom“researchonresearch”studiesinorthodontics(in

italics).7-11