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Repair of bilateral cleft lip and nose by the Mulliken method: a case report

Jae-Seok Lim1, Gyu-Tae Lee1, Young-Soo Jung1,2

1Department of Oral and Maxillofacial Surgery, 2Oral Science Research Center, College of Dentistry, Yonsei University, Seoul, Korea

Abstract (J Korean Assoc Oral Maxillofac Surg 2012;38:360-5)

The simultaneous surgical correction of bilateral cleft lip and nasal deformity has become a more common surgical technique that has greatly changed conventional strategies for secondary nasal correction. Mulliken has been known as one of the earliest proponents for the synchronous repair of bilateral cleft lip and nasal deformity, and he emphasized the responsibility of the treating surgeon to evaluate nasolabial growth by comparing anthropometric measurements with age-matched normal patients. Good outcomes from this surgical method have been reported in clinical cases worldwide. Herein, we describe the management of two cases of bilateral cleft repair, following the principles and methods established by Mulliken. We also provide a relevant review of the literature.

Key words: Bilateral cleft lip, Nasolabial growth, Four-dimensional change, Synchronous repair[paper submitted 2012. 2. 16 / revised 2012. 5. 9 / accepted 2012. 5. 13]

sideredtobemuchmoredifficultthantreatmentofunilateral

cleftlip3,andthesurgicaltechniquehasgraduallydeveloped

fromthestagedclosureapplyingunilateralcleftlipsurgeryto

simultaneousclosure2,4.Inthepast,attentionwasfocusedon

thelipitself,andcorrectionofnasaldeformitywaspostponed

becauseearlymanipulationofnasalcartilagewasthought

toprevent itsgrowth.Such thought,however,hasbeen

foundtobegroundlessbyresearch5,andsynchronousrepair

ofbilateralcleftlipandnosedeformitywasintroduced,in

whichtreatmentofbilateralcleftlipwasaccompaniedwith

correctionofnasaldeformity.

Mulliken isapioneerofsynchronousbilateralcleft lip

andnasaldeformityrepair.Heestablishedthefollowingfive

surgicalprinciplesin1985:1)maintenanceofsymmetry;2)

establishmentofbasicmusclecontinuity;3)propersizeand

shapeofthephiltrum;4)formationofthemediantubercle

fromthelaterallipelements;and5)primarypositionofthe

alarcartilageforconstructionofthenasaltipandcolumella.

Mullikensaiditisimportanttounderstandandapplythese

surgicalprinciplesratherthanthesurgicaltechniqueitself,

andthroughresearchongrowthmeasurementsofbilateral

cleft lippatients,healsoemphasized the importanceof

takinggrowthintoconsideration2,6.Authorsaretoreporta

caseofapplyingtheMulliken’sprinciplesandtechniquesfor

treatmentofbilateralcleftlippatients.

I. Introduction

Cleftsoflipandpalatearethemostcommondeformities

inoralandmaxillofacialarea.Althoughdifferentresearch

institutesinKoreareportedvariousincidenceratesregarding

cleftsoflipandpalate,theyaregenerallyknowntoshowan

incidenceof0.95-2.25per1,000newbornbabies1.Bilateral

cleftlip,inparticular,maybesymmetricalorasymmetrical

duetoitscomplexaspects.Itappearsinvariousformsfrom

thecaseofcleftliponlytothecaseofcleftlipaccompanied

withalveolarcleftorcleftplate.Moreover,thesizes,shapes

ofitsgrowthsegmentsandtheratiosbetweenthesegments

changealongwith thegrowthof thepatient.Therefore,

successful surgical repairofbilateral cleft lip requires

understandingof thesevariousaspectsandsuchchanges

occurringalongwiththegrowthofthepatient2.

Treatmentofbilateralcleft liphasbeenclassicallycon-

Young-Soo JungDepartment of Oral and Maxillofacial Surgery, Oral Science Research Center, College of Dentistry, Yonsei University, 50, Yonsei-ro, Seodaemun-gu, Seoul 120-752, KoreaTEL: +82-2-2228-3139 FAX: +82-2-2227-7825E-mail: YSJOMS@yuhs.ac

This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

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CASE REPORThttp://dx.doi.org/10.5125/jkaoms.2012.38.6.360

pISSN 2234-7550·eISSN 2234-5930

Repair of bilateral cleft lip and nose by the Mulliken method: a case report

361

to immoderatedissectionandclosure ifsurgeryhadbeen

performedwithoutdealingwiththeproblemfirst.Therefore,

afterpremaxillaosteotomy,premaxillawas repositioned

backward toa favorablepositionforsurgery.(Figs.2,3)

Afterwards,surgerywasperformedintheorderofmarking,

dissection,closureandnasalcorrection.Sincethesurgeries

wereperformedaspartofourvolunteerserviceactivities

overseas,itwasunabletocarryoutalong-termobservation

ofthepatients’clinicalcoursepostoperatively.

1. Flap marking

Thephiltralflapwasdesignedtobealittlebiconcaveand

II. Case Report

WevisitedHanoiNationalHospitalofOdonto-Stomatology

in2009and2011,andthesameoperatorperformedsurgery

ontwobilateralcleft lipandnosepatientsonthebasisof

Mulliken’sprinciplesandtechniques.Thefirstpatientwas

an18-month-oldmale,whowasobservedtohaveprotrusive

premaxilla because early surgical repair hadnot been

performed.(Fig.1.A)Thesecondpatientwasan8-month-

oldmale,whodidnothavesevereprotrudingpremaxilla.

(Fig.1.B)Inthecaseofthefirstpatient,itwasdifficultto

restorealveolarridgecontinuityduetoprotrusivepremaxilla,

excessivesofttissuetensionwouldhavebeenexpecteddue

Fig. 1. Preoperative clinical photograph. A. 18 months child with protrusive premaxilla. B. 6 months child without protrusive premaxilla.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 2. Intraoperative clinical photograph (18 months child). A. Osteotome was applied to posterior of epiphysial line. B. Premaxillary osteotomy was done.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:360-5

362

2. Dissection

Thephiltral flapwas incisedupto thedermis, thenthe

piecesofskinonitssideweredeepithelized,andthenthe

remainingprolabiallipskinwasexcised.Thephiltralflap,

includingthesubdermalsoft tissue,waselevatedfromthe

premaxilla to thecaudal septum.Analarbase flapwas

formedbyincisingthelateralwhiteline-vermilion-mucosal

flap,andabundleoforbicularisorismuscleswasdissected

fromthelaterallabialelements.(Fig.5)

3. Closure

Thealveolarridgeonbothsidesofthecleftwasincised

verticallyandthenclosedbyraisingthegingivomucoperiosteal

itsendtobedart-shapedbecauseofthetendencyofcicatrix

tobulgeoutwardlaterontheedgesofitsbothsides.Along

narrowstripwasdesignedonbothsidesofthephiltralflap

fordeepithelization,whichwasthenperformedinorderto

formaphiltralridgebyplacingitbelowthelaterallabialflap.

ThepeaksoftheCupid’sbowtobeformedwereplacedon

thelateral labialelements.Analarbaseflapwasdesigned

on theupper junctionof the lateral labialelements.The

medialedgesof the lateral labialelementsweredesigned

alongrightabovethevermilion-cutaneousline.(Fig.4)The

secondpatienthadaleftnostrilsill,butsincethesillwas

unnecessaryforadvancementofthealarbaseflap,anincision

linewasalsoestablishedonit.(Fig.4.B)

Fig. 3. Surgical repositioning of premaxilla (18 months child). A. Protrusive premaxilla was observed. B. Premaxilla was moved backward.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 4. Marking. A. 18 months child. B. 6 months child.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 5. Dissection of the orbicularis oris muscle and elevation of flap.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Repair of bilateral cleft lip and nose by the Mulliken method: a case report

363

4. Nasal correction

Thebilateralalarcartilagewasexposedbyincisingthe

bilateralnostrilrim.Interdormalmattresssuturewasperfor-

medonthegenuasofthebilateralalarcartilageaswellason

itsmiddlecruses,andthenthesuturedgenuasandthesutured

middlecrusesweresuturedtotheupperlateralcartilageatthe

lateralsideofgenuaandatthelateralcrusrespectively.The

alarwidthwasnarrowedto25mmorbelowbypositioning

thecinchsutureoneachalarbase.(Fig.6)Anostrilsillwas

constructedbycuttingandrefiningtheendofanalarbaseflap

andthenrotatingitinward,thussuturingittotheendofthe

skinonbothsidesofthecolumellarbase.Thedermisofeach

alarbaseflapwassuturedtotheperiosteumofthepremaxilla

andtheorbicularisorismuscle,thusforminganormalcymal

shapeofthelateralsillandloweringthepositionofthealar

base.Thiscouldminimizethenostrilsbeingliftedduringthe

patient’ssmilebystimulatingthedepressoralaenasimuscle.

(Fig.7)

Ifalarcartilageisproperlypositioned,therewillclearly

beextraskininthesofttriangle.Theextraskinwasexcised

togetherwiththeremainingskinonthelateralsideof the

columella.Crescenticexcisionwasextendedtothetopand

middleof thecolumella,passing the incision lineon the

nostriledge.Moreover,alateralvestibularwebappearsinside

thenose.Thiswebwasincisedbylenstypeincisionalongthe

intercartilaginousline,afterwhichitwassutured.(Fig.7)

Aftercompletionofnasalcorrection,thelastskinclosure

wasperformed.Theendofthephiltralflapwasplacedinside

andsuturedtothemuscularlayerinordertoformaphiltral

ridgebyhaving thephiltral flapsettledown.(Fig.8)The

flap.Theanteriorwallof thegingivolabial sulcuswas

shortenedbyexcisingandrefining thevermilion-mucosa

remaininginpremaxilla.Theremainingmucousmembraneof

thepremaxillawasmadetobeaflap,whichwasthensutured

totheperiosteumoftheanteriornasalspine,thusachieving

formationoftheposteriorwalloftheanteriorgingivolabial

sulcus.Byadvancingthelaterallabialelementfullyinward,

medialmucosalflapsweremadetoformtheanteriorwallof

thecentralgingivolabialsulcusandtheposterioraspectofthe

upperlip.Theorbicularisorismusclesweresuturedfromthe

bottomtothetop.Theparsperipheralis,thetopclosure,was

suturedtotheperiosteumoftheanteriornasalspine.(Fig.5)

Themediantuberclewasformedusingtheremainingportion

ofthelaterallip.Thelastskinclosurewasperformedafter

nasalcorrection.

Fig. 6. Narrowing interalar dimension with cinch suture.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 7. Inferior view after suturing. A. 18 months child. Cymal trim was done at superior edge of lateral labial flap. B. 6 months child.Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:360-5

364

Aspremaxillaryosteotomywasperformedintheposterior

partoftheepiphyseallineandthesurgicalrepositioningof

thepremaxilla tookplacewithoutanyparticularfixation,

wethinkthegrowthofthebabypatient’smidfacewouldbe

inhibitedonlyslightly.

Itisnecessarytounderstandthenasalandlabialshapeofa

normalbabyforcorrectionofbilateralcleftlip,butonlythree-

dimensionalunderstanding isnotsufficient.Thesurgeon

mustbeawareof thechanges thatwouldappearduring

thenormalgrowthandevenbeable topredictdistortions

occurring in the patientwho had underwent surgery.

Accordingly,it isnecessarytoperformfourth-dimensional

treatmentbytakingchangesingrowthintoconsideration.

Mullikenetal.10confirmedthatthereisacorrelationbetween

thethree-dimensionalsizeimmediatelyafterthesurgeryand

thegrowthspeedbygrowthobservationofbilateralcleft

lippatientswhohadunderwentsurgery. Inotherwords,

thefast-growingpartswouldgrowtobe longerorwider

whereastheslow-growingpartswoulddeformtobeshorter.

Thesurgerymustmakethefast-growingpartssmallerand

theslow-growingpartsslightlybiggerthannormal.Farkas

etal.11mentionedintheirgrowthresearchinnormalwhite

peoplethatnasaltipprotrusionandcolumellarlengthgrow

slowlywhilereportingthatallthelabialpartsgrowfast,thus

reaching90%ofanadult’slabialpartssizebytheageof5

years.Alsointhetwocases,surgerywasperformedbased

onthesegrounds,thusnarrowingthenasalwidthtobe25

mmorbelowandincreasingthecolumellarlength.Sincelips

growfastexceptthemediantubercle,thepatient’slipswere

formedtobeshorterandnarroweronanoverallbasis.(Fig.8)

It isknown that therearevariousmethodsofprimary

heightofthelaterallipwasmodifiedbycuttingandrefining

thetopedgeofthelaterallabialflapintheformofacymal

curveatthealar-labialjunction.(Fig.8.A)

III. Discussion

Thesuitabletimeforperformingprimarycheiloplastyon

bilateralcompletecleftlippatientsmaybevariableaccording

to literatures,but it isgenerallyperformedbetween3 to

6monthsafterbirth7.Incasesofprotrusivepremaxilla, it

isnecessary to treat it surgicallyornonsurgicallybefore

primarycheiloplasty.Sothetimingofprimarycheiloplasty

maybeadjustedaccordingtothepriorsurgicalornonsurgical

treatmentof theprotrusivepremaxilla.Protrusionof the

premaxillaisaphenomenonappearingduetotheabnormal

growthof theboneunder the influenceof thestrengthof

stickingoutthetonguelocatedinthebilateralcleftliptogether

withtheabnormalmuscularstrengthoftheorbicularisoris

muscleswhicharenotcontinuousdueto thecleft lip8. In

thefirstcase,surgicalrepositioningofthepremaxillawas

performedon the first18-month-oldpatient (Fig.1.A)

becauseitwasimpossibletoachieveaprimaryrepairwithout

treatingthepremaxillafirst.Surgicalrepositioningof the

premaxillaisrecommendedfor6to8-year-oldpatientsrather

thanyoungerpatientsbecausesurgical repositioningmay

hamperthegrowthofthemidface9.Inthefirstcase,however,

premaxillaryosteotomywasperformedonthepatientduring

thecourseofprimarycorrectionofcleftlipandnasaldeformity

despitehisageof18monthsbecauseneitherperiodical

observationofhisclinicalcoursenoruseofanorthopedic

appliancewaspossibleduetoeconomiccircumstances.(Fig.2)

Fig. 8. Frontal view after suturing. A. 18 months child. B. 6 months child. Jae-Seok Lim et al: Repair of bilateral cleft lip and nose by the Mulliken method: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Repair of bilateral cleft lip and nose by the Mulliken method: a case report

365

patientswerealsoorientalpeopleinthiscase,weappliedthe

modifiedthree-dimensionalpositiontothesurgicaloperation.

Theprinciplesoftheprimarycorrectionofbilateralcleft

lipandnasaldeformityhadbeenestablished,andthesurgical

techniqueisgraduallydeveloping.TheMullikenmethodisa

four-dimensionaltreatmenttakinggrowthintoconsideration,

andgoodresultsarebeingreportedbothinKoreaandabroad

throughthissurgicalmethod6,16.Wehopethiscasereportbe

helpfulfororalandmaxillofacialsurgeonsinapplyingthe

Mulliken’streatmentmethodintheirsurgicaloperations.

References

1. BaikHS,KeemJH,KimDJ.Theprevalenceofcleft lipand/orcleftpalateinKoreanmaleadult.KoreanJOrthod2001;31:63-9.

2. MullikenJB.Bilateralcleftlip.ClinPlastSurg2004;31:209-20.3. BrownJB,McDowellF,ByarsLT.Doublecleftsofthelip.Surg

GynecolObstet1947;85:20-9.4. MillardDR.Bilateral cleft lip andaprimary forked flap: a

preliminaryreport.PlastReconstrSurg1967;39:59-65.5. SalyerKE.Primarycorrectionoftheunilateralcleftlipnose:a15-

yearexperience.PlastReconstrSurg1986;77:558-68.6. MullikenJB.Bilateralcompletecleftlipandnasaldeformity:an

anthropometricanalysisofstaged tosynchronousrepair.PlastReconstrSurg1995;96:9-23.

7. PreciousDS,GooddayRH,MorrisonAD,DavisBR.Cleftlipandpalate:areviewfordentists.JCanDentAssoc2001;67:668-73.

8. FigueroaAA,ReisbergDJ,Polley JW,CohenM. Intraoral-appliancemodificationtoretractthepremaxillainpatientswithbilateralcleftlip.CleftPalateCraniofacJ1996;33:497-500.

9. PadwaBL,SonisA,BagheriS,MullikenJB.Childrenwithrepairedbilateralcleftlip/palate:effectofageatpremaxillaryosteotomyonfacialgrowth.PlastReconstrSurg1999;104:1261-9.

10. MullikenJB,BurvinR,FarkasLG.Repairofbilateralcompletecleftlip:intraoperativenasolabialanthropometry.PlastReconstrSurg2001;107:307-14.

11. FarkasLG,PosnickJC,HreczkoTM,PronGE.Growthpatternsofthenasolabialregion:amorphometricstudy.CleftPalateCraniofacJ1992;29:318-24.

12. MullikenJB.Principlesandtechniquesofbilateralcompletecleftliprepair.PlastReconstrSurg1985;75:477-87.

13. MillardDRJr.Columella lengtheningbya forked flap.PlastReconstrSurgTransplantBull1958;22:454-7.

14. CroninTD.Lengtheningcolumellabyuseofskinfromnasalfloorandalae.PlastReconstrSurgTransplantBull1958;21:417-26.

15. McCombH.Primaryrepairofthebilateralcleftlipnose:a15-yearreviewandanewtreatmentplan.PlastReconstrSurg1990;86:882-9.

16. KimSK,LeeJH,LeeKC,ParkJM.Mullikenmethodofbilateralcleft liprepair:anthropometricevaluation.PlastReconstrSurg2005;116:1243-51.

cheiloplastyforbilateralcleftlip,butnoneofthemisperfect.

Traditionalmethods,inparticular,involvepullingthemedial

cruraofthenosebackwardanddownward,thuscausingthe

columella tobeshorter12.Becauseof these limitationsof

surgicalmethods,mostsurgeonsperformprimarycorrection

whilekeeping inmindsecondarysurgery forcolumellar

lengthening.Millard13 andCronin14 stored some forked

flapsintheinferiorpartofthenasalsillduringtheprimary

surgeryand thenused themin thesecondarysurgery. In

1990,however,McComb15reportedunaestheticresultssuch

asnasal tip lengthening,unnaturalnostrils,andexcessive

lengtheningof thecolumella in their retrospectivestudy

of thetwo-stagesurgeryfor15years.Mullikenalsoused

the two-stagesurgeryfornasalcorrection initially.After

understanding,however,thatthenoseofabilateralcleftlip

andnosepatientjustlooksshortbutisnotreallyshort,he

startedtoperformnasalcorrectionwithoutanyadditional

tissue transplant. Inaddition,heestablished thepresent

surgicalproceduresafterstoppingnasaltipverticalincision

inordertominimizecicatrixes.Hereportedthatasaresultof

retrospectivecomparisonofhissurgicalprocedures,thetwo-

stagesurgeryandtheone-stagesurgerygenerallyshowed

similarresults.Alsointhetwocases,nasalcorrectionwas

performedafteranapproach throughthenostrilmarginal

incisionwithoutusingthenasaltipverticalincisionapproach.

(Fig.7)

Inprimarycorrectionofcleftlipandnasaldeformityusing

theMullikenmethod, the three-dimensionalpositionwas

determinedbasedonthegrowthofwhitepeople.Mulliken

proposedthatthelengthofthephiltralflapbe6-8mm,that

thedistancebetweenthepeakofCupid’sbowbe3-4mm,

andthatitshouldbe2mmatthecolumellar-labialjunction.

Kimetal.16,however,announcedthebilateralcleftlipsurgery

performedonsomeKoreansusing theMullikenmethod,

alsomentioningthenecessityofsomemodificationinthe

three-dimensionalpositionduetothetendencyofKoreans

havingfewertissuesthanwhitepeople.Theyproposedthat

thelengthofthephiltralflapbeaslongaspossible,thatthe

distancebetweenthepeakofCupid’sbowbe5mm,andthat

itshouldbe3mmatthecolumellar-labialjunction.Sincethe