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Abstract Inthecontemporaryera,thedemandfororthodontictreatmentiseverrising.Orthodontictreatmentdurationcanrangefromayeartoafewyears.Ouraimistoassesstheavailabletechniquesof categorising treatment effectiveness in patients with cleft lip and palate (CLP) and to studytheireffecton improvementof treatmentoutcomes.Theelectronicdatabases includingMedline-PUBMED,ScienceDirect,andISIWebofKnowledgeweresearchedfrom1987to2013,and40311relevantarticleswerefound.Ofthese,weidentified22articlesincludingoriginalarticlesaswellasliteraturereviews.Thedifferentparametersandindicesthatareappliedtospeed-uporthodontictreatment outcomes in patients with CLP were identified as the GOSLON Yardstick, 5-year-oldindex,EUROCRANindex,HuddartBodenhamsystem,modifiedHuddartBodenhamsystem,GOALYardstick and, Bauru-Bilateral Cleft Lip and Palate Yardstick. This overview can create betterawarenessregardingtheuses,advantages,anddisadvantagesofthedifferentindices.ItcanenablebetterassessmentandprovidetheimpetusneededforasustainedupgradeinthestandardsofcareforCLPindailyorthodontics.

Keywords: cleft lip and palate, malocclusion, treatment outcome, orthodontic index

Introduction

There are several types of congenitalcraniofacial anomalies, most frequent of whichare orofacial clefts that encompass the cleft lipandpalate(CLP),whichoccurswhenembryonicfacialprocessesfailtounite(1).Thecomplicationsassociated with CLP are maxillary growthaberrations and high occurrence of Class IIImalocclusions.InchildrenwithCLP,aberrationsin number, size, shape, and period of toothformationaremorecommonthaninthenon-cleftpopulation. Orthodontic abnormalities such ascrowding, rotation,andmalpositionof teetharealsofrequentinpatientswithCLP(2). ‘Intheorthodonticcontext,anindexisusedtodesignatearatingorasacategorisingsystemthat assigns a numerical score or alphanumericlabeltoaperson’socclusion’(3). Indiceshavebeendeveloped formeasuringtheoutcomeoftreatmentmorepreciselyinordertodeterminethedegreeofsuccessintreatingthecleftdefects.Anidealmeasureofoutcomeshouldbeeasytolearn,quicktoapply,reliable,andvalid.There are different types of indices that assesstreatment outcome in patients with CLP, suchasthefollowing:GOSLONYardstick(4);5-year-

old index (5); EUROCRAN index (6); HuddartBodenham system (7), and Modified HuddartBodenham(mHB)system(8,9). The aim of this overview is to identify andassess the different indices that are used forcategorising the treatment outcome in patientswithCLP.Further,weaimtoanalyse,frompreviousstudies, theeaseofuseofanyspecific index, itsreliability,validity,andextentofapplication.Ourstudy also investigates the compatibility of theindices with statistical scrutiny. It will benefitclinicians in the selection of a specific index forscoring the treatment outcome and enable thereview of treatment options to ensure betterpatientcare.

Methods and Materials

Inviewoftheimportanceofdifferentindicesin CLP in orthodontics, a 2-examiner-basedsearchinliteraturewasconducted.Theelectronicdatabases searched from 1987 to 2013 includedMedline-PUBMED,ScienceDirect, and ISIWebof Knowledge search engines, from which 40311 articles were included in the study initially.Referencesofrelevantarticleswerethensearchedmanuallyand22articleswerefinallychosen,after

Review Article An Overview of Indices Used to Measure Treatment Effectiveness in Patients with Cleft Lip and PalateSanjida Haque, Mohammad Khursheed alam, Anas Imran arsHad

Orthodontic Unit, School of Dental Science, Universiti Sains Malaysia, Health Campus, 16150 Kubang Kerian, Kelantan, Malaysia

Submitted:15Jul2014Accepted:4Nov2014

4Malays J Med Sci. Jan-Feb 2015; 22(1): 4-11

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applying the selection criteria. The language ofthe articles was restricted to English. Originalresearch articles as well as literature reviewswere selected. The selection criteria includedthe following: appropriate quantity of subjects,quality of data assessed, typeof cleft treatment,scoringsystemused,statisticalanalysesused,andthe conclusions reached. The following free-textterms were used for the searches: Cleft lip andpalate, Cleft indices, Crossbite index, GOSLONindex, 5-year-old index, Huddart-Bodenhamindex,EUROCRANindex,andmBH.

Results

The results of the literature survey fordifferentindicesinrelationtoCLPareshownintable1.

Discussion

The different types of indices along with theiruses,advantages,anddisadvantagesarediscussedbelow:

GOSLON Yardstick TheGreatOrmondStreet,LondonandOslo,Norway (GOSLON) Yardstick was developedfor categorising the degree of malocclusion(maxillary growth) with unilateral cleft lip andpalate (UCLP). The GOSLON Yardstick wasintroducedbyMars et al. (4).Contrastingothersystems, the GOSLON Yardstick is treatment-linked(e.g.anteriorcrossbitewithretroclinationoftheincisorscanbecorrectedmoreeasilythananteriorcrossbitewithnormalincisorinclination)and is therefore more useful than a specificanomaly-score alone. Not only the enucleatingeffect but also the hereditary skeletal pattern isaddressed by this scoring system, as it is basedon the prospects for orthodontic rectification.The system was developed for categorising thedegree of malocclusion in 10-year-old childrenwith UCLP, examined in the late mixed orearly permanent dentition (4). It categorisesmalocclusionsinpatientswithUCLPaccordingtoantero-posteriorarch,verticallabialsegment,andtransverserelationships.Uses:

● It is useful in the assessment of dentalrelationshipsinUCLP

● It has been developed for use in the latemixedandearlypermanentdentition

● It is valuable in predicting treatmentneed (orthodontic treatment, surgicaltreatment)

Advantages:● TheGOSLONYardstickhasproven tobe

able of discriminating arch relationshipsand interference of facial morphologyoutcomesbetweendifferentcentres(10)

● Itconsidersclinically importantvariablesin all 3 planes of space and permits therankingofmodelsintheorderofdifficultytoachieveafavourableoutcome(11)

● Ithasbeenshowntohavegoodinter-andintra-examinerreliability(12)

● Ithasbeenverifiedasaneasyandpracticalevaluation to differentiate between thequalitiesofdegreeofmalocclusionduringallstagesofdentaldevelopment(10)

● Itcanpredictsurgicaloutcomesatanearlyageof5years(5)

Disadvantages: The GOSLON Yardstick is an ordered andcategorical classification, which is expectedto be less powerful than an objective constantnumerical measurement scale. Moreover, acontinuous scale measurement more eagerlysatisfiestheassumptionsofparametricstatisticalanalysis(11). The GOSLON Yardstick requires thejudgestobetrainedintheuseof this indexandrecalibration is necessary to assure consistency(11). The GOSLON Yardstick can only be usedtoscoreUCLPandnoothercleft types(11).Thevalidity of the GOSLON Yardstick has not beeninvestigatedanditispredictedtobedifficultsinceitrequiresaclusterofadultswithUCLPwhohavebeentreatedbyprimarysurgeryonly(11).

5-year-old index Developed by Atack et al. (5) to overcomethe shortcomings of the GOSLON Yardstick,this index assesses studymodels of 5-year-olds.It allows surgeons to assess their treatmentoutcomesmorepreciselysothattheycanenhancetheirclinicalskills.Uses:

● ItassessesdentalrelationshipsinUCLP● Ithasprimarilybeendevelopedfor5-year-

oldpatients

Advantages:● Itisamorereliabletoolinmeasuringstudy

models of 5-year-olds than the GOSLONYardstick(13)

● This index has been shown to haveexcellent intra-examiner and good inter-examinerreliability

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Table1:LiteraturesurveyofdifferentindicesinrelationtocleftlipandpalateAuthor UsedIndex OutcomeSusamietal.(17) GoslonYardstick ResultshowedgoodreproducibilityHsiehetal.(18) GoslonYardstick ProvidedreliabletreatmentoutcomeChanetal.(19) GoslonYardstick Showednosignificantgroupdifferencein

themodelscoresofthetwogroupsKajiietal.(20) GoslonYardstick ShowedreliableresultsMorrisetal.(21) GoslonYardstick ProvidedusefulbaselinedataMarsetal.(22) GoslonYardstick SufficientlyreliableforgeneraluseAltalibietal.(23) GoslonYardstick

5yearoldindexEurocranindexHuddart-BodenhamModifiedHuddart-bodenham.GoalYardstickBauru-BilateralCleftLipandPalateYardstick

ModifiedHuddart–BodenhamIndexequaledoroutperformedthebestamongallindices

GOSLONYardstickwasthemostcommonlyusedindexduetoalongertimeinuse

Hathronetal.(24) GoslonYardstick. ShowedreliableresultsLiljaetal.(25) GoslonYardstick ProducedthebestGOSLONYardstick

ratingsAlametal.(2) 5-year-oldindex

GoslonYardstickGotsatisfactoryresultsbyusing5-year-oldindexandGoslonYardstick

Patel(11) EurocranYardstick.ModifiedHuddart-Bodenham(mHB).

mHBismorereliablethanEurocranYardstick

Flinnetal.(26) 5-year-oldindex ShowedexcellentreliabilityDibiaseetal.(27) 5-year-oldindex Suitabletoolforassessingtheoutcomeof

treatmentSuzukietal.(28) 5-year-oldindex

Huddart-BodenhamindexOcclusaloutcomeofcaseswithUCLPwasfairasevaluatedusingthe5-year-oldindex

Clarketal.(29) 5-year-oldindex ShowedreliableresultsHathornetal.(30) 5-year-oldindex Outcomeswereimprovedcomparedwith

previousnationaloutcomesJohnsonetal.(31) 5-year-index ProvidedafavorableoutcomeFudaljetal.(32) Eurocranindex ShowedreliableoutcomeFudaljetal.(6) Eurocranindex TreatmentoutcomewasreliableGrayandMossyetal.(2005)(16)

ModifiedHuddart-BodemhamsystemGoslonYardstick5-year-oldindex

ResultshowedthatthemodifiedHuddart/BodenhamsystemprovidesanobjectiveandreliabilityAssessmentofmaxillaryarchconstriction

Dobbynetal.(2011)(17)

ModifiedHuddart-BodemhamsystemGoslonYardstick5-year-oldindex

ModifiedHuddart-Bodenhamhadbeenshownamuchsensitivescoringsystem

Koshikawa-Matsunoetal.(2014)(33)

GoslonYardstick5-year-oldindexDentalmodelanalysis

Regardingbothindices,nosignificantdifferenceswerefound.However,thedentalarchwidthshowedsomesignificantvariations

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Disadvantages:● Truevalidationofthisindexisnotpossible

anditreliesonfacevalidity● Like the GOSLON Yardstick, the 5-year-

oldindexisalsoordinal● Thisindexisnotversatile● The examiners need to be calibrated;

therefore, it iscomplextouse forscoringsurgicaloutcomes

● Itcanbeusedonlyin5-year-oldpatients

EUROCRAN Yardstick The EUROCRAN Yardstick index wasdevelopedbytheparticipantsoftheEUROCRANproject (2000–2004). This project was anextensionoftheEUROCLEFTprojectwiththeaimto recover research capabilities. This index wasdevelopedbyusingfindingsfromtheassessmentof a mix of 118 cases from different Europeancentres. A tally using the GOSLON Yardstickand the 5-year-old index had been maintainedforthesecases.Thescoresshowedthatonlyoneofthecaseswasgradedas5,andtwocasesweregradedas 1byall theexaminers involved in thestudy.Therefore,owingtotheredundancyoftheextremesinthescaleof1to5,itwasdecidedthatthegradeoptionsbereducedto4intheantero-posterior, vertical, and transverse dimensions,insteadofthe5-gradescale.Inaddition,a3-gradescalewasallocatedforratingthepalatalform. Thus, the EUROCRAN Yardstick is amodification of the GOSLON Yardstick and5-year-old index, and it is again designed toassesssurgicaloutcomes inpatientswithUCLP.It is applied to study models, and the majorcomponents of this index include the degree ofmalocclusionintheantero-posteriorandverticaldimensions,andthepalatalform.

Uses:● Itisusefulinassessingsurgicaloutcomes

inpatientswithUCLP● Itcanbeappliedtoevaluatethedegreeof

malocclusioninbothantero-posteriorandverticaldimensions,aswellasthepalatalform

Advantages:● The supremacyof theEUROCRAN index

isitsvalidity(6)● Inordertoamplifyitsjudiciouspower,the

indexhasadiscretepositionfordegreeofmalocclusionandpalatalmorphology

● ComparedtotheGOSLONYardstick,thisindex gives a more meticulous guide forcataloguingoftreatmentconsequences

● Ithasbeenshowntohavemoderatetoverygoodinter-andintra-examinerreliability

Disadvantages:● Itrequireselaboratestudy● It is difficult to apply and relies on

conjectures. Consequently, there is moreroomforerror

● Therearetoomanydetailstoconsider,andtoomanypreconditionsandmodifications

● It is more time-consuming and is moredifficulttolearnthanthemHB

● Scoringthepalatalvaultisdifficult● Scoringthepalatalvaultissubjective(11)

Huddart Bodenham system The original Huddart Bodenham scoringsystemwasdevelopedin1972.Ithas5categoriesfor scoring incisors and 3 categories for scoringcaninesandmolars.

Uses:● This system is used in the assessment of

treatmentoutcomesinpatientswithUCLP● Itcanbeappliedinpatientswithdeciduous

teeth● It isuseful inpatientsbelowtheageof6

years

This system was devised subsequent toevaluation of 2 other categorical indices, whichwere devised by Pruzansky and Aduss, andMatthewsetal.,bothofwhichassessthepresenceand degree of crossbite, both anteriorly, andposteriorly.Thestudyconcludedthatboththesecategorical indices were not consistent in thehands of different observers, because categoriesincludedsharpdelineationandsharpdelineationsdo not extend to occlusion. As a result, a greatdeal of subjective judgment was required tobe employed and information regarding theindiceswasunreliableindifferenthands,sinceitwouldbeverycomplicated toestablishcommonassessmentcriteria.Thepaperalsodeclaredthatemploying two different indices, which were sodifferent, made effective comparison of resultsbetween centres very difficult. Therefore, theauthorsattemptedtodevisean indexwhichwasnumerical, gavemore detailed information, andlentitselftostatisticalanalysis(14,15).

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PruzanskyandAdussdividedtheocclusioninto6categories(14):

● Nocrossbite● Caninecrossbiteonly● Buccalcrossbiteonly● Anteriorandbuccalcrossbite● Anteriorandcaninecrossbite● Incisorcrossbiteonly

Incontrast,Matthewsetal.dividedtheocclusionasfollows(15):

ClassA:themaxillaandthemandibleareinidealocclusionwithallsegmentsClassB(1):thetoothborderingthecleftonthelessersegmentisinlingualocclusionClassB(2):normalocclusionofthegreatersegmentbutlingualocclusionofthelessersegmentClassB (3): themaxillaryarch isperfect,butistoosmallClassC:themaxillaisnotonlyinclassIIIpositionwith all segments, but there is acollapse of a number of fractions of thesmallmaxillaryarch

The mHB system This scoring system was developed afterconsidering the above disadvantages. Thissystem is described as ‘modified’ because itwasdeveloped fromtheoriginalHuddartBodenhamindex.Mosseyetal.,aswellasGrayandMosseycomparedthisindexwiththeGOSLONand5-yearold indices. The comparisons showed the mHBsystem to be more reliable, objective, sensitive,andsimpletouse(8,9).

Uses:● Itmeasuresmaxillaryarchconstrictionin

patientsbornwithUCLP● Itisapplicableinanytypeofcleft● Itmeasures severity of the crossbite and

each maxillary tooth is scored accordingtoitsrelationshipwiththecorrespondingtoothinthemandible.

Advantages:● It is more versatile in that this index is

applicableatanyageafter3yearsand inanytypeofcleft

● Itismorereliable,objective,andsensitivethantheGOSLONand5-year-oldYardstickindices(9)

● Itissimpletouse

Disadvantages:● It does not score for antero-posterior

skeletal and vertical discrepancies,and does not take into account incisorinclinations(16)

● Thisscoringsystemhasbeenvalidatedonstudymodelsonly

Different authors have studied differentindices to obtain varying results. Susami et al.examinedstudymodelsof24patientswithUCLP,all prior to orthodontic treatment and alveolarbone grafting. The GOSLON Yardstick wasused to rate the degree of malocclusion. Intra-and inter-examiner agreements estimated byweighted kappa statistics were high, indicatinggoodreproducibility(17). The degree of malocclusion was evaluatedviatheGOSLONYardstickusingintraoraldentalphotographs. These data suggest that intraoraldentalphotographsdeliveratrustworthymethodforratingthedegreeofmalocclusion(18). Inanotherstudy,non-syndromicCaucasianchildrenwithUCLPweredivided into2groups.Patients of age ranging from5 to 10 years,whohad been treated either with or without activeinfant orthopaedics, were selected. The studydid not find any significant disparity betweenthe two groups. While the orthopaedic groupdemonstratedameanGOSLONscoreof3.30,thenon-orthopaedicgroupscored3.21(19). Kajii et al. obtained all the necessaryinformation from plaster models and assessedthesamebyusingtheGOSLONYardstick.Theirresearch pointed to requisite intra- and inter-examineragreements,whichwasassessedusingweightedkappastatistics(20). Morris et al. assessed themaxillary growthinchildrenbornwithacompleteUCLPbetween1983 and 1987, who had undergone primarycleftrepair.ThetreatmentoutcomeofthisUCLPsample was then compared with the results ofpreviously published articles. The models wereassessed by using the GOSLON Yardstick. Theresultswereofaslightlyhigherstandardthanthatofpreviouslypublishedarticles(21). Mars et al. categorised malocclusions inpatientswithUCLPinawaythatwouldsymbolisetheseverityofmalocclusionandthedifficulty incorrecting it. The results of the study exhibitedthat the GOSLON Yardstick was highly reliableandwasdiscriminatingofthequalityoftreatmentresults(22).

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In a very recent literature review articleaboutdifferent indices thatareused tomeasurethetreatmenteffectivenessinpatientswithUCLP,the GOSLON Yardstick was stated as the mostfrequently used index and themHB as the bestexecuted index, according to the WHO criteria(23). Hathron et al. assessed 32 study modelsof patients with UCLP by using the GOSLONYardstick. More than 50% of the sample wasin the unfavourable GOSLON Groups IV andV. Hathron et al. planned their next surgicaltreatmentprotocolbasedonthisassessment(24). Lilja et al. found that at 19 years of age,85%ofthepatientswithUCLPwereinGOSLONGroups I and II, whereas 12%were assigned toGroupIII.Only3%ofthecaseswerefoundtobeinGroupIV.NodentalstudymodelwasfoundtobeinGroupV.Thisexceptionallongitudinalstudyof patients with UCLP demonstrates the bestdegreesofmalocclusionthusfarpresentedusingtheGOSLONYardstick(25).In another study, the consequence of maxillarygrowth by applying the 5-year-old index andGOSLONYardstickwas examined. Itwas foundthat 69% and 79% of subjectswere confidentialinto the favourable group using the 5-year-oldindexandGOSLONYardstick,respectively(20). Patel compared the reproducibility of themHBandEUROCRANYardstick.Sheexamined30studymodelsbyusingthesetwoindicesandthestudyrevealedthatthemHBismorereliablethantheEUROCRANYardstick(11).Flinn et al. analysed 118 consecutively treated5-year-oldpatientswithcomplete,non-syndromicUCLP.Average ratings of dental casts using the5-year-old Yardstick was computed for eachpatient and results showed excellent reliability(26). Dental arch dimensions of children in theprimary dentition with repaired UCLP werecompared with that of a non-cleft group of asimilar age by Dibiase et al., using the 5-year-oldindexTheresultsshowedthatthe5-year-oldindex was an appropriate device for evaluatingtheeffectsof treatment intheprimarydentitionforantero-posteriorandanteriortransversearchdimensions(27). Both 5-year-old and Huddart Bodenhamindices were compared by Suzuki et al. in theevaluation of dental arch dimensions. Resultsshowed that the occlusal outcome of caseswithUCLPwas fairasevaluatedusing the5-year-oldindex(28).A similar study was carried out to evaluate theproper utilisation of the 5-year-old index. The

selectedmodelshadbeenmadebetweenMay1992andApril1998,andonlypatientswithUCLPwereincluded in the study. Two qualified examinersmeasured the study models twice by using the5-year-oldindex,andtheindexdemonstrateditsworthconclusively(29). Hathorn et al. also used the 5-year-oldindex to gauge study models. They found animprovementintheresultsascomparedtoearliernationaltreatmentoutcomes(30).Johnsonetal.studiedthegradingofthedegreeofmalocclusioninstudymodelsusingthe5-year-oldindex. The inter- and intra-examiner agreementkappa statistics revealed good to very goodagreementusing this index,andthis indicatedafavourableoutcome(31).Fudaljetal.comparedthedegreeofmalocclusionfollowing 1-stage and 3-stage surgical protocolsforUCLP.Theyanalysed61dentalcastsusingtheEUROCRAN Yardstick and the results showedreliableoutcomes(32). Fudalj et al. again studied the degreeof malocclusion in 2 groups—exposed andunexposed—with UCLP that had been operatedbythesamesurgeon.Thedegreeofmalocclusionand palatal morphology were rated separatelyby using the EUROCRAN Yardstick, and thetreatmentoutcomewasfoundtobereliable(6). A study was undertaken to appraise thecomparisonoftheeffectivenessofthemHBsystemwiththatofthe5-year-oldandGOSLONindicesin subjects with UCLP. Reiterated assessmentwas performed after a 1-month interval by 4appraisers. It was found that the mHB systemgave a credible valuation of the maxillary archconstriction(9). A similar studywas performed using studymodelsofsubjectswithUCLPfromEnglandandScotland. All the models had been previouslyscoredby applying the 5-year-old andGOSLONindices.Themodelswerere-evaluatedbyapplyingthe mHB system to compare the consequencesand themHB index proved to be amuchmoresensitivescoringsystem(16). Koshikawa-Matsunoetal.recentlyconductedastudyon74patientswithUCLP,andtheyusedtheGOSLONYardstick in combinationwith the5-year-old index and dental model analysis. Byapplyingweightedkappaanalysis,theyconcludedthattherewassufficientinter-andintra-examineragreement(33). Thus,wehave revieweddifferent indicesofdiversenature in relation tocleft lipandpalate.Asystematicreviewofsuchcomplexindicesmayleadtobetterassessmentandcontrollerbias.

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Conclusion

From this study, it can be concluded thatdifferent indices like the GOSLON Yardstick,5-year-old Yardstick, EUROCRAN Yardstick,Huddert-Bodenham index, and mHB index areusefultoolsinclinicalorthodonticsformeasuringtreatment effectiveness in patients with CLP.The GOSLON Yardstick is the most commonlyusedindex.ThemHBindexisencouragingintheassessment ofmalocclusions related to all typesofCLPofallagesandinregulatingtheextentofoutcomesinpatientswithCLP.TheEUROCRANYardstickisafavouritebecauseitcanbeusedtoevaluatethedegreeofmalocclusioninbothantero-posteriorandverticaldimensions,aswellas thepalatal form. The 5-year-old index is the idealindexfor5-year-oldpatients.Inorthodontics,theuseofacombinationofdifferenttypesofindicesappearstobebeneficialandpromising.

Acknowledgement

None.

Conflict of Interest

None.

Funds

None.

Authors Contributions

Conception and design, drafting of the article,critical revision of the article for the importantintellectualcontent,finalapprovalof thearticle,collectionandassemblyofdata:SH,MKA,AIA

Correspondence

DrMohammadKhursheedAlamBDS(DU),PGT(DU),PhD(Japan)OrthodonticUnitSchoolofDentalScienceUniversitiSainsMalaysiaHealthCampus16150KubangKerianKelantan,MalaysiaTel:+6014-2926987Fax:+609-7642026Email:dralam@gmail.com dralam@usm.my

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