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François GIRINON, Serge KETOFF, Quentin HENNOCQ, Nicolas KOGANE, Nicolas ULLMANN,
Natacha KADLUB, Eva GALLIANI, Cécilia NEIVA-VAZ, Marie-Paule VASQUEZ, Arnaud PICARD,
Roman Hossein KHONSARI - Maxillary shape after primary cleft closure and before alveolar bone
graft in two different management protocols: A comparative morphometric study - Journal of
Stomatology, Oral and Maxillofacial Surgery - Vol. 120, n°5, p.406-409 - 2019
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Maxillary
shape
after
primary
cleft
closure
and
before
alveolar
bone
graft
in
two
different
management
protocols:
A
comparative
morphometric
study
F.
Girinon
a,
S.
Ketoff
a,b,
Q.
Hennocq
b,
N.
Kogane
b,
N.
Ullman
b,
N.
Kadlub
b,
E.
Galliani
b,
C.
Neiva-Vaz
b,
M.P.
Vazquez
b,
A.
Picard
b,
R.H.
Khonsari
b,*
a
Artsetme´tiersParisTech,LBM,Paris,France b
AssistancepubliqueHoˆpitauxdeParis,HoˆpitalUniversitaireNecker-EnfantsMalades,ServicedeChirurgieMaxillofacialeetChirurgiePlastique,Centrede Re´fe´rencedesMalformationsdelaFaceetdelaCavite´ Buccale(MAFACE),Filie`reMaladiesRaresTeteCou,Universite´ ParisDescartes,Universite´ SorbonneParis Cite´,Paris,France
1. Introduction
Thescientificassessmentofmanagementprotocolsforcleftlip andpalateraisesnumerousmethodologicalissues.Theseprotocols coverlong periodsof time –from birthto theend of pubertal growth– and many controllable and non-controllable factors influencethesurgicaloutcomes,suchas,amongmanyothers,the choice of the surgical techniques, the timing of the different procedures,theskillsofthevarioussurgeonsinvolved,detailsin
thepost-operativecare,thequality oftheorthodontic manage-mentandspeechtherapytechniques[1–3].
ThemaxillofacialandplasticsurgerydepartmentatNecker -EnfantsMaladesUniversityHospitalisaveryactivenationalcleft centermanaging200newprimarycaseseveryyear.In2012,the departmentchangeditssurgicalteamandmanagementprotocol forchildrenwithcleftlipandpalate. Until2012,protocolNo.1 involved an early closure of thelip at the age of 1–3months followedbyacombinedclosureofthesoftandhardpalateatthe ageof6–9months.Bonegraftforalveolarcleftreconstructionwas notpartofprotocolNo.1.From2012,protocolNo.2wasapplied: thesoftpalateandthelipwereclosedat6monthsofage,thehard palatewasclosedat18monthsofageandthealveolarbonegraft wasperformedbetweenages4 and6,usinganiliacdonorsite. Alveolarcleftreconstructionwasprecededbytheplacementofan
Keywords: Cleftlipandpalate Geometricmorphometrics Cone-Beam
Maxillarygrowth Functionalprotocol
ABSTRACT
Aimandscope:Resultassessmentincleftsurgeryisatechnicalchallengeandrequiresthedevelopmentof dedicatedmorphometrictools.Twocohortsofpatientsmanagedaccordingtotwodifferentprotocols wereassessedatsimilaragesandtheirpalatalshapewascomparedusinggeometricmorphometrics. Materialandmethods:Tenpatients(protocolNo.1)benefitedfromearlylipclosure(1–3months)and secondarycombinedsoftandhardpalateclosure(6–9months);11patients(protocolNo.2)benefited fromlatercombinedlipandsoftpalateclosure(6months)followedbyhardpalateclosure(18months). Cone-BeamComputedTomography(CBCT)imageswereacquiredat5yearsofageandpalatalshapes werecomparedbetweenprotocolsNo.1andNo.2usinggeometricmorphometrics.
Results:ProtocolsNo.1andNo.2hadasignificantlydifferenttimingintheirsurgicalstepsbutwere assessedatasimilarage(5years).Theinter-caninedistancewassignificantlynarrowerinprotocol No.1.Geometricmorphometricsshowedthatthepremaxillaryregionwaslocatedmoreinferiorlyin protocolNo.1.
Conclusion:Functionalapproachestocleftsurgery(protocolNo.2)allowobtaininglargerinter-canine distancesandmoreanatomicalpremaxillarypositionsat5yearsofagewhencomparedtoprotocols involvingearlylipclosure(protocolNo.1).Thisisthefirststudycomparingtheintermediateresultsof twocleftmanagementprotocolsusing3DCBCTdataandgeometricmorphometrics.Similarassessments attheendofpubertyarerequiredinordertocomparethelong-termbenefitsoffunctionalprotocols.
* Corresponding authorat: Servicede Chirurgie Maxillofaciale etChirurgie Plastique, Hoˆpital Universitaire Necker-Enfants Malades, 149, rue de Se`vres, 75015Paris,France.
intra-oralorthopedicexpansiondevice(Quad-Helix)andthegraft wasperformedwhentheinter-caninedistancewasover32mm, beforetheeruptionofthepermanentincisorsandcanines[3].
Inordertocomparetheoutcomeofcleftlipandpalateclosure betweenprotocols No.1 andNo.2, weretrospectivelyincluded thelastseriesofpatientsinitiallymanagedaccordingtoprotocol No.1andincludedintoprotocolNo.2attheageof4(in2012), wheneligibleforalveolarcleftreconstruction.Wecomparedthe 3D structure of their maxilla based on Cone-Beam Computed Tomography(CBCT)imageswithaseriesofpatientsfullymanaged according to protocol No.2 since birth. Using morphometric geometrics [4,5], we assessed the differences in maxillary structuresbetweenthetwogroups.
2. Materialandmethods
Weincluded10patientsmanagedaccordingtoprotocolNo.1 andincludedintoprotocolNo.2at4yearsofage(groupNo.1), and 11 patients initially managed according to protocol No.2 (group No.2), without agenesis of the lateral incisors. We consideredCBCTsperformedbeforeorthopedicmaxillary expan-sion. We recorded the following parameters: age at primary surgery,ageatsecondarysurgery,ageatCBCTandinter-canine distance (measured on 3D reconstructions obtained from the CBCTs).
We defined three anatomical regions on the surface of the palate:
1. palataltip(inred),extendingtoahorizontallinedistaltothe canines;
2. medianpalatalregion(inwhite),coveringtherestofthehard palatedowntothemaxillo-palatinesuturesand;
3. posteriorpalate(ingreen),coveringthesurfacebetween the maxillo-palatinesuturesandtheposteriorchoana(Fig.1).
Apalatalplanewascomputedinordertofitthepalatalsurface usingtheleast-squaremethod.Thez-axiswasdefinedasnormalto thispalatalplane.Thebarycentreofthepalataltipwascomputed andprojectedonthepalatalplane.Thebarycentreoftheposterior palatewascomputedandsimilarlyprojectedonthepalatalplane. Thex-axiswasdefinedusingthesetwoprojections.The displace-mentvectorsbetweenthetwogroupswereprojectedintothese axes.Weconsideredthefirst5principalcomponents,accounting for80%ofthevariance,afterhavingtestedthemulti-normalityof thedatasetusingaHenze–ZirklertestfromtheMVNpackage[6]in R (R Development Core Team (2008). R: a language and environmentforstatisticalcomputing.RFoundationforStatistical Computing, Vienna, Austria. ISBN 3-900051-07-0, URL http:// www.R-project.org).Amultivariateanalysisofvariance(MANOVA test) for each individual in the space of the first 5 principal componentswasperformed.
3. Results
GroupNo.1consistedin7boys(6left-sidedcleftsand1right sidedcleft)and3girls(2left-sidedcleftsand1rightsidedcleft). GroupNo.2consistedin7boys(6left-sidedcleftsand1rightsided cleft)and4girlswithleft-sidedclefts.ThemeanagesatCBCTwere notdifferentbetweenthetwogroups:5.951.01yearsforgroup No.1and5.090.97forgroupNo.2(P=0.06).Theagesatprimary surgeryweredifferentbetweenthetwogroups:1.920.96months forgroupNo.1and5.091.16monthsforgroupNo.2(P<0.001). Theagesatsecondarysurgerywerealsodifferentbetweenthetwo groups:8.41.22monthsforgroupNo.1and15.644.59months forgroupNo.2(P<0.001)(Table1).Theinter-caninedistancefor groupNo.1was2.560.32,andwassignificantlyshorterthanthe inter-canine distance for group No.2, which was 2.900.28 (P=0.026).
Fig.1.Definitionofthetotalregionofinterestonthesurfaceofthepalate(inred),andofthethreedifferentzonesusedinthemorphometricanalyses:(1)palataltip(red),(2) medianpalatalregion(white)and(3)posteriorpalate(green).
Table1
ClinicalcharacteristicsofgroupsNo.1andNo.2.
Sexratio(m/f) Cleftside(L/R) MeanageatCBCT(y) Ageatprimarysurgery(mo) Ageatsecondarysurgery(mo)
GroupNo.1 7/3 8/2 5.951.01 1.920.9 8.41.22
GroupNo.2 7/4 10/1 5.090.97 5.091.16 15.644.59
DistancesbetweenpalatalsurfacesfromgroupsNo.1andNo.2 were computed for the whole palate, the palatal tip and the posteriorpalate(Table2).TheMANOVAtestinthespaceofthefirst 5 principal components showed that the 2 groups were significantly different for the position of the palatal tip, while the rest of the palatal surface was superimposable (Table 3). Graphical rendering of these results showedthat the different betweengroupsNo.1andNo.2waspredominantlylocatedinthe tipofthehardpalate,inthepremaxillaryregion.
4. Discussion
Variousmorphometricmethodshavebeenusedtoassesscleft anatomy[7–13]andtheoutcomesofcleftrepair[14,15],basedon 2D radiographic data [7,8], 3D radiographic data [9,10] and digitizeddentalcasts[11,15].Ourstudyisneverthelessthefirst,to ourknowledge, to use3D CBCTdata in order to compare the outcomes of two different management protocols based on geometricmorphometrics.
Protocol No.1 involved early lip closure (at
1.920.96months)andsecondarycombinedsoftandhardpalate closure (at 8.41.22months); protocol No.2 was based on functionalconsiderations[3,16,17,18]andfavoredlaterlipclosure associatedwithsoftpalateclosureusingintra-velarveloplasty(at 5.091.16months) followed by hard palate closure (at 15.644.59months). Protocol No.2 allowed minimizing palatal scars and could potentially favor transversal andsagittal palatal growth[3,19,20].Here,weshowedthattheinter-caninedistanceat 5years of age was significantly smaller in protocol No.1, thus suggestingthatthefunctionalapproachtocleftsurgerycouldfavor transversal maxillary growth [20]. This transversal effect was nevertheless not confirmedbygeometric morphometrics(Table3
andFig.2)andmostprobablyrequiresassessmentsbeyond5yearsof age. Interestingly, we showed that, at 5years of age, the main differencesbetweenthepalatalshapesinprotocolsNo.1andNo.2 werelocatedatthepalataltip,inthepremaxillaryregion.Thisfinding wasinlinewithpreviousresultsfromtheliterature,whichreported aninferiordisplacementofthepremaxillaincleftpatients[12,13];
Table2
DisplacementsbetweengroupsNo.1andNo.2(incentimeters).
Wholepalate Palataltip Posteriorpalate
x y z x y z x y z
Meandisplacement 0.25 0.03 0.09 0.36 0.13 1.03 0.14 0.05 0.22
Minimalabsolutedisplacement 0 0 0 0 0 0 0 0 0
Maximalabsolutedisplacement 1.01 0.82 1.73 0.73 0.74 1.73 0.5 0.77 0.65 Meanofthedisplacementnormstandarddeviation 0.560.34 1.150.29 0.400.18
Fig.2.DisplacementsbetweengroupsNo.1andNo.2accordingtothex-,y-andz-axes,showingthatthedifferencesinshapepredominatedatthetipofthepalate,inthe premaxillaryregion.Foreachdisplacedpoint,theroundendrepresentedgroupNo.2:thesignificantdownwarddisplacementofthepalataltipingroupNo.2wasthus figuredbyapositive(inredandyellowcolorcodes)downwardshiftoftheroundendsofeachindividualdisplacementalongthez-axis.ProtocolNo.2furthermoreinduceda trendforposteriortransversalnarrowing(y-axis,notsignificant);thesagittaldimension(x-axis)wasnotsignificantlyaffectedat5yearsofage.
Table3
MANOVAtestsshowingthatthepositionofthepalataltipregionissignificantlydifferentingroupsNo.1andNo.2intheprincipalcomponentspace.
df P F df1 df2 p
Whole palate 1 0.29 2.09 5 26 0.09
Palatal tip 1 0.34 2.68 5 26 0.04
Posterior palate 1 0.09 0.52 5 26 0.75
Df: degree of freedom; P: Pillai’s trace; F: Fisher F-test; df1, df2: degrees of freedom used in determining the F statistics; significant results in red.
basedonthesepreviouslypublishedresults,wesupportthefactthat functionalprotocolNo.2allowsobtainingapalatalshapecloserto normalthanprotocolNo.1.
5. Conclusion
Comparingtheresultsoftwocleftmanagementprotocolsasa wholeisanimpossibletask.Byconsideringdiscretestepswithin theseprotocols,andbyfocusingonspecific3Dobjectsandlimited morphometric parameters (here: 3D palatal shape), we could demonstratea significantly differentpalatal structure resulting fromtwoapproachestocleftsurgery:
earlylipclosure;
functional approach involving lip and soft palate closure at 6monthsofage[3,19].
Thecausesofthesedifferencescannotbededucedfromour results;itisneverthelessgenerallyhypothesizedthatlipsurgeryat 6monthsofageallowsabetteranatomicalreconstructionthatin early surgery, and that a two-step palatal closure minimizes scarring[20].Thesetwofactorsmostprobablyinfluencepalatal growth,andmorepreciselypremaxillarygrowth;interestingly,the premaxillary region is the only significantly modified region betweenprotocolsNo.1andNo.2accordingtoourresults.Our findings areobtained at an intermediateage. Longerfollow-up assessmentswouldprovidemoredefinitiveargumentsinfavorof the use of a functional approach in cleft lip and palate management.
Disclosureofinterest
Theauthorsdeclarethattheyhavenocompetinginterest.
Acknowledgments
ThankstoPr.Ge´rardCoulyandDr.BenoıˆtMichelforsharing datafromtheirpatients.
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