160

Partial sublingual glandectomy with ranula excision: a new conservative method for treatment

In-Kyo Chung1, Hyo-Ji Lee1, Dae-Seok Hwang1, Yong-Deok Kim1,

Hae-Ryoun Park2, Sang-Hun Shin1, Uk-Kyu Kim1, Jae-Yeol Lee1

Departments of 1Oral and Maxillofacial Surgery, 2Oral and Maxillofacial Pathology, School of Dentistry, Pusan National University, Yangsan, Korea

Abstract (J Korean Assoc Oral Maxillofac Surg 2012;38:160-5)

Objectives: This study evaluated the clinical results of partial sublingual glandectomy accompanying the excision of ranula as new treatment modality.Materials and Methods: A total of 43 patients who were treated between 1999 and 2007 for oral or plunging ranula were reviewed. All patients were treated surgically by various methods with a total of 55 different procedures performed. Ten cases of partial sublingual glandectomy with excision of the ranula were conducted. All excised specimens were examined. We compared the clinical outcomes resulting from each treatment method.Results: The recurrence rates for marsupialization, excision of ranula, marsupialization with gauze packing, total excision of sublingual gland and ranula, and partial sublingual glandectomy with excision of ranula were 50%, 25%, 25%, 0% and 10%, respectively. Of the 10 patients treated by partial sublingual glandectomy with ranula excision, only one experienced recurrence (10%), i.e., plunging ranula. None of the ranulas contained an epithelial lining, and the excised portion of the feeding sublingual glands showed degenerative changes.Conclusion: In removal of ranulas, we found that excision of the attached sublingual gland, which removed the feeding portion and degenerative acinar cells, yielded good outcomes. Thus, as a new conservative method for treatment, we recommend partial sublingual glandectomy to accompany excision of the ranula.

Key words: Ranula, Sublingual gland, Treatment outcome[paper submitted 2012. 2. 6 / revised 2012. 3. 15 / accepted 2012. 3. 16]

ofRiviniandWhartonductshavebeenreported1.Inthiscase,

a trueepithelial liningmadeof theepitheliumoftheduct

wasdetected.Whenaranulaspreadsoutfromtheposterior

inferiorareaof themylohyoidmuscle to the submental

areaorcervicalarea, it isknownasaplungingranula.In

general, ranulasappearasabluishdome-shapedswelling

onthefloorofthemouth.Whenmucocelesaccumulatein

thesubcutaneousareaorneartheepidermisarea,aranula

appearsasalightbluishcolorbecauseofepidermalcyanosis

andretainedsalivainthesubcutaneousarea.Whenaranula

existsinthedeeperareas,however,themucosaappearsasan

ordinarypinkishcolor.

Aranuladoesnotcauseserioussymptomsofpainexcept

somediscomfort, and ithardlygives rise toanysevere

clinicalmanifestation.According toBaurmash1,clinical

findingssuchasdiscomfort in speech,mastication,and

swallowingandexternalswellingdifferdependingonthe

sizeandlocationoftheranula.Inthecaseofaverylarge

mucoceleinthesublingualgland,thetonguemaycompress

I. Introduction

Aranula is amucoceleoccurringon the floorof the

mouthandisderivedeitherfromtheextravasationofsaliva

outof thesalivaryductsor retentionofsaliva inside the

salivaryducts,originatingintheminorsalivaryglandorthe

sublingualglandasoneof themajorsalivaryglands1,2. In

general,aranulaisknownasanextravasationphenomenon

ofthesublingualgland3-6.Itlookslikeacysticlesion,butit

isapseudocystwithoutepitheliallining3,7-12.Note,however,

thattherehavebeenfewcaseswhereinmucusretentioncysts

Jae-Yeol LeeDepartment of Oral and Maxillofacial Surgery, Pusan National University Hospital, 179 Gudeok-ro, Seo-gu, Busan 602-739, KoreaTEL: +82-51-240-7432 FAX: +82-51-231-7429E-mail: id9753153@gmail.com

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.

CC

ORIGINAL ARTICLEhttp://dx.doi.org/10.5125/jkaoms.2012.38.3.160

pISSN 2234-7550·eISSN 2234-5930

Partial sublingual glandectomy with ranula excision: a new conservative method for treatment

161

52surgical treatmentswereperformedonthe43patients.

(Table1)Mucoceles inareasother than the floorof the

mouthandoccurrencesontheWharton’sductwereexcluded.

Basedonthemedicalrecords,weinvestigatedthediagnosis,

treatment,statusofrelapse,andcomplications.Theresults

ofeachsurgicalprocedurewerereviewed,andweassessed

theclinicalresultsofpartialsublingualglandectomywith

theexcisionof the ranula incomparisonwith theother

methodsused.Histologicalexaminationswereperformedon

allexcisedspecimens.Eachranulawascompletelyexcised,

exceptinthecaseofmarsupialization.Thefollow-upperiod

wasfromatleastsixmonthsuptotwoyears.

Among the43patients, 10were treatedwithpartial

sublingualglandectomywithexcisionoftheranula.Surgery

wasperformedundergeneralanesthesia.Ellipticalincision

wasmadeonthemucosaof theranula.Meticulous,blunt

dissectionwasperformedalongwiththepseudocystupto

thesublingualgland.Whendissectionreachedthebodyof

thesublingualglandsecretingtheranula,thelocationofthe

attachmentportionof thesublingualglandwasconfirmed

andexcisedfor1-1.5cm.(Fig.1)Theresectedmarginofthe

sublingualglandwassuturedwith4-0non-absorbablesilk,

andtheincisedmucosawasclosedusingageneralmethod.

Theotherproceduresused-exceptpartialglandectomy-

werecarriedoutusingordinarymethods.

III. Results

Atleast39and4patientswerediagnosedwithranulason

thefloorof themouthandplungingranulas,respectively.

theranuladuringeatingandswallowingsuchthat thereis

interferencewith thesalivaryflowof thesubmandibular

gland.Whenaplunging ranula increases insize, itmay

causedyspneaanddysphagiaandmayexpandasfarasthe

mediastinum13.Thereareseveralsurgicalmethodsfor the

treatmentofranulas,withvaryingcurerate7,8,14.Although

asimpletechnique,marsupializationhasbeenreportedto

haveahighrecurrencerate.Tominimizeanyrecurrence,

Baurmash2proposedmarsupializationwithgauzepacking

andsuggestedthattheremovaloftheentiresublingualgland

couldbeovertreatment insomecases. Incontrast, there

havebeenseveral reportsof theextirpationof theentire

sublingualgland,recordingarelativelyhighcurerate.Thus,

theremovalof theentiresublingualgland isproposed to

minimizeanyrecurrence7,8,11,14-16.Note,however, that the

completeremovalofthesublingualglandmayinvolvethe

functionaldeteriorationofthesalivaryglandsandincrease

thepossibilityofdamage to theWharton’sduct, lingual

nerves,andbloodvessels6,16.

Inthisresearch,wehaveinvestigatedtheusefulnessofa

procedureinvolvingthesimultaneouspartialremovalofa

damagedareaofthesublingualglandwiththeexcisionofthe

ranula.Thismethodmaintainsthefunctionofthesublingual

glandandminimizestherecurrencerate.Here,wedescribe

partialsublingualglandectomywiththeexcisionoftheranula

asanewtreatmentmodelandcompareitwithothersurgical

procedures.

II. Materials and Methods

Retrospectiveevaluationwasperformedon43consecutive

patientsdiagnosedwith ranulasduring theperiod1999-

2007at theauthors’ institution.Thepatients’agesranged

from3to75years,withaverageageof21years.Atotalof

Table 1. Surgical methods and recurrence rates

OperationtypeNumberof

operationcasesNumberofrecurrences

Recurrencerate(%)

ExcisionofranulaMarsupializationMarsupializationwithgauzepackingTotalexcisionofsublingualglandandranulaPartialsublingualglandectomywithexcisionofranulaTotal

24104

4

10

52

651

0

1

13

25.050.025.0

0

10.0

25.0

In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conser-vative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 1. Partial sublingual glandectomy with excision of ranula.In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conser-vative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:160-5

162

ranula,andtheconditionwasresolved.

Inonecaseofexcisionoftheranula,thepatientcomplained

ofparethesiaontheipsilateralsideofthetongueimmediately

afterthesurgery,butthesymptomswereresolvedtwoweeks

laterwithnofurther treatment.Other thanpost-operative

swellingordiscomfort,noothercomplicationswerefound.

Accordingto thehistological tests,nolesionswith true

epithelial liningwerefound.Themucuscavitywas lined

withlooseconnectivetissueandgranulationtissue.These

showedfibroustissue,inflammatorycells,foamyhistiocytes,

andlymphocytesintheconnectivetissue.Degenerationof

aciniccells frompartiallyexcisedsublingualglandsand

dilationoftheductwereobserved.(Fig.3)Inflammationand

degenerationweredetectedinthesublingualglandadjacent

totheranula.

IV. Discussion

Ranulaisaclinicaltermdenotingamucoceleoccurringon

thefloorofthemouth.Theetiologyofaranulaisdiscussed

Therewere24casesofexcisionoftheranula,10casesof

marsupialization,4casesofmarsupializationwithgauze

packing,4casesoftotalexcisionofthesublingualglandand

ranula,and10casesofpartialsublingualglandectomywith

excisionoftheranula.Therecurrencerateswere25%,50%,

25%,0%,and10%,respectively.

Eightpatientsunderwentpartialsublingualglandectomy

withexcisionoftheranulaasthefirstoperation,andoneof

thepatientssufferedfromrecurrence.(Table2)Thismale

patientwas3yearsoldandwasdiagnosedwitharanulaat

thefirstvisit.Onemonthafterthesurgery,thepatienthada

recurrentlesion.Thepatientwasrediagnosedwithaplunging

ranulaandsubsequently treatedwith totalexcisionof the

sublingualglandandranula.

Intwoofthepatientswhowereeachtreatedwithexcision

oftheranulaandmarsupializationinthefirstsurgery,ranulas

recurredonemonthlater.Thesepatientswereoperatedon

usingpartialsublingualglandectomy(Fig.2),andneither

sufferedfromrecurrence.Therecurrentlesionsweretreated

withpartialsublingualglandectomywithexcisionof the

Table 2. Partial sublingual glandectomy with excision of ranula

Case Gender/age Dx. Site 1stoperation 2ndoperation1 Result

12345678910

Male/3Male/5Male/6Male/8Female/8Female/8Female/14Female/75Female/22Female/30

PlungingranulaRanulaRanulaRanulaRanulaRanulaRanulaRanulaRanulaRanula

Lt.Lt.Rt.Rt.Lt.Rt.Rt.Rt.Rt.Lt.

PSGPSGPSGPSGPSGPSGPSGPSGEM

TENNNNNNNPSGPSG

N

NN

(Dx.:diagnosis,Lt.:left,Rt.:right,PSG:partialsublingualglandectomywithexcisionofranula,E:excisionofranula,M:marsupialization,TE:totalexcisionofsublingualglandandranula,N:norecurrence)1inrecurrencecasesafterthe1stoperation.In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conservative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 2. Gross findings of excised ranula and excised portion of sublingual gland. Solid arrow: ranula, hollow arrow: atta-ched sublingual gland.In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conservative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

Partial sublingual glandectomy with ranula excision: a new conservative method for treatment

163

thelesionisalsosmall8.Thecommonformofaranulaisa

pseudocystderivedfromtheextravasationofthesublingual

gland3-17.Thereasonfortheextravasationisconsideredto

betraumatothesublingualglandandrelatedstructure1,7,8.

in termsofductalobstructionanddamageof thesalivary

duct17.The retentionofmucusandductaldilationoccur

withtheobstructionofthisduct.Theresultisanepithelial-

linedcyst.Observationof thesecases is rare; thesizeof

Fig. 3. Histopathologic features of ranula. A, B. A cystic cavity is surrounded by loose connective tissue or granulation tissue (H&E staining, A: ×50, B: ×120). Adjacent feeding sublingual gland (arrow) is seen near the cavity. C. The cystic wall is composed of numerous inflammatory cells, foamy histiocytes, lymphocytes, and fibrous tissue (H&E staining, ×150). D, E. In the sublingual gland, acinic cells show degenerative change (H&E staining, ×400). F. Ducts (arrows) are dilated and degenerated (H&E staining, ×500). In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conservative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:160-5

164

of thesublingualgland.Baurmash5 insists that the total

excisionofthesublingualglandisnotappropriateforsmall-

sizedranulasandsuggestsamodifiedmarsupializationwith

gauzepacking.Baurmashused thismethodon12cases,

with1caseofrecurrencereported.AccordingtoBaurmash,

however,marsupializationcannotresolveranulasassociated

with thedeeppartof the sublingualgland. Inaddition,

marsupialization(eitheronceorrepeated)cancausefibrosis

oftheupperside;thus,mucuscanbeinducedinthelower

partofthelesion,resultinginaplungingranulaincaseof

recurrence15.

Tosummarizetheviewpointof thehealingprocess, the

previouslyreviewedtreatmentmethodcanbecategorized

asfollows:chronicfistulizationofthelesion;sealingofthe

feederbase;excisionof theranulaonly,and; removalof

thesublingualsalivaryglandastheorigin.Marsupialization

formsachronicfistulabetween themucussecretionpart

andtheoralcavity.Note,however,thatmostranulasdonot

haveatrueepithelial lining;thus,achronicfistulacannot

beestablished8,11.Consequently,thereisrecurrencewiththe

healingoftheoralepithelium.Thetechniqueofsealingthe

feederbaseinducesatrophyandfibrosisofthedamagedpart

ofthesublingualgland.Marsupializationwithgauzepacking,

cauterizationusingCO2laser20,21,andinjectionofpicibanil(OK-

432)22,23fallunderthiscategory.Marsupializationaccompanied

bygauzepackingmaintainsachanneltotheoralcavityand

allowstimeforfibrosisofthemucussecretionarea;henceits

lowerrecurrenceratethansimplemarsupialization.Similarly,

fibrosisandsclerosisofthemucussecretionpartareinduced

byCO2laserandinjectionofOK-432.Inthistrial,however,

thepossibilityofrecurrencestillexistsbecausethedamaged

partof thesublingualglandisnotexcised.Theprocedure

fortheexcisionoftheranulaonlyremovesthecyst,soithas

ablindpoint,i.e.,itcanmissthefeedergland.Theremoval

ofthesublingualglandasthesourceofthepseudocystisa

radicaltreatment;henceitspotentialcomplications.

Partialsublingualglandectomywithexcisionoftheranula

Whentheaciniccellsaredamagedbytrauma,salivaleaks

into adjacent tissueand formsa cystic cavity linedby

granulation tissuesurroundedby looseconnective tissue.

Manystudieshaveconfirmedthattheexcisedranulahasno

trueepithelial lining3,7-11.Theetiologyforaranula isstill

notclear,however1,18.Inthisresearch,nolesionswithtrue

epithelialliningwereobserved;thus,ranulasarethoughtto

beanextravasationphenomenonofthesublingualgland.

Therehavebeenseveralmethodsusedforthetreatment

ofranulas,butpropermodalityisstillasubjectofdebate

becauseofthetendencytowardrecurrence.Therecurrence

ratesinliteraturearesummarizedinTable3,decreasingin

thefollowingorder:marsupialization,excisionofranula,and

excisionofthesublingualgland.Zhaoetal.14statedthatthe

recurrenceratesofranulaswerenotrelatedtothepatternsof

lesionbutwerecloselyrelatedtothemethodofthesurgical

procedureused.Crysdaleetal.8suggestedexcisionof the

sublingualgland for ranulas>1cm.Catoneet al.11 and

Bridgeretal.15proposedexcisionofthesublingualglandas

theprimarytreatmentforallranulasregardlessofsize.

Becauseof its simplicityandcomplicationsafter the

excisionof thesublingualgland,manycliniciansprefer

marsupializationas thefirst treatment16,19.Complications

associatedwith thesurgicalmanagementof ranulashave

beeninvestigatedbyZhaoetal.16.Accordingtothem,the

mostfrequentcomplicationsaretherecurrenceoflesionand

numbnessofthetongueduetodamagetothelingualnerve.

Damage to theWhartonduct,hemorrhaging,hematoma,

wounddehiscence,andinfectionhavebeenreportedaswell.

Recurrencewasfrequentinthemarsupializationandexcision

of theranula.Injurytoadjacent tissuewasrelatedmainly

totheexcisionofthesublingualgland.Note,however,that

thesecomplicationsafter theexcisionof theranulawere

temporaryandwereresolvedwithinagiventimeframe.Thus,

tominimizerecurrenceasthemostcommoncomplication,

theremovalofthesublingualglandhasbeensuggested.

Somecliniciansarestillskepticalastothetotalremoval

Table 3. Recurrence rates (%) and number of cases inliterature

I&D M MP E ESG

Crysdaleetal.8

Yoshimuraetal.7

Zhaoetal.14

ChidzongaandMahomva19

100(4)---

61(19)36.4(22)66.67(9)

20(10)

0(2)---

0(4)25(4)

57.69(26)-

0(11)0(9)

1.05-1.55(415)0(73)

(I&D:incisionanddrainage,M:marsupialization,MP:marsupializationwithgauzepacking,E:excisionofranula,ESG:excisionofsublingualgland)Valuesarepresentedas%(number).In-Kyo Chung et al: Partial sublingual glandectomy with ranula excision: a new conservative method for treatment. J Korean Assoc Oral Maxillofac Surg 2012

Partial sublingual glandectomy with ranula excision: a new conservative method for treatment

165

TetsumuraA,etal.MRIofranulas.Neuroradiology2000;42:917-22.

4. ShelleyMJ,YeungKH,BowleyNB,SneddonKJ.Ararecaseofanextensiveplungingranula:discussionofimaging,diagnosis,andmanagement.OralSurgOralMedOralPatholOralRadiolEndod2002;93:743-6.

5. BaurmashHD.Marsupializationfor treatmentoforalranula:asecondlookat theprocedure.JOralMaxillofacSurg1992;50:1274-9.

6. BaurmashHD.Treatingoralranula:anothercaseagainstblanketremovalof the sublingualgland.Br JOralMaxillofacSurg2001;39:217-20.

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8. CrysdaleWS,MendelsohnJD,ConleyS.Ranulas--mucocelesof theoral cavity: experience in26children.Laryngoscope1988;98:296-8.

9. GallowayRH,GrossPD,ThompsonSH,PattersonAL.Pathogenesisandtreatmentofranula:reportofthreecases.JOralMaxillofacSurg1989;47:299-302.

10. MoritaY,SatoK,KawanaM,TakahasiS,IkarashiF.Treatmentofranula--excisionofthesublingualglandversusmarsupialization.AurisNasusLarynx2003;30:311-4.

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12. BarnesL.Surgicalpathologyoftheheadandneck.1sted.NewYork:MarcelDekker;1985:1297-301.

13. MarxRE,SternD.Oralandmaxillofacialpathology. Illinois:QuintessencePublishing;2003:511.

14. ZhaoYF,JiaY,ChenXM,ZhangWF.Clinicalreviewof580ranulas.OralSurgOralMedOralPatholOralRadiolEndod2004;98:281-7.

15. BridgerAG,CarterP,BridgerGP.Plungingranula: literaturereviewandreportofthreecases.AustNZJSurg1989;59:945-8.

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17. QuickCA,LowellSH.Ranulaandthesublingualsalivaryglands.ArchOtolaryngol1977;103:397-400.

18. HarrisonJD,GarrettJR.Anultrastructuralandhistochemicalstudyofanaturallyoccurringsalivarymucoceleinacat.JCompPathol1975;85:411-6.

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cansatisfymanyoftherequirementsdiscussedabove.First,

itremovestheranulalesionaswellastheoriginofthelesion

byexcisingthemucus-supplyingpartofthesublingualgland.

Suturingof theexcisedmarginof thesublingualglandis

intendedtosealthefeederbase.Furthermore,thesublingual

glandhasseveralsecretionductsthatopenindependently;

thus, functional recoveryof thesublingualglandcanbe

expectedevenafter theexcisionofpartof thesublingual

gland.Inaddition, it isnoninvasivecomparedtothetotal

extirpationofthesublingualgland,andsoitisexpectedto

havefewercomplications.

Partialglandectomyismainlyapplied tooperationson

theparotidgland,andsuperficialparotidectomyispracticed

widely.Partialsuperficialparotidectomyhasbeenintroduced

as amoreconservativemethod than superficialparoti-

dectomy24.Thispartialsalivaryglandectomyisbasedonthe

regenerativeabilityofthesalivaryglands,andthehealing

procedureofdamagedsalivaryglandsofrodentshasbeen

reported25,26.Note,however,thattherehavebeennoreports

onpartialsublingualglandectomy.Similartotheregeneration

ofparotidglands, sublingualglandsareexpected togo

throughasimilarhealingprocedure.

V. Conclusion

Inthisstudy,wehaveshownthatpatientswhounderwent

partialsublingualglandectomywiththeexcisionofaranula

recoveredwithoutanynoticeablecomplicationandwithlow

recurrencerate.

Therefore,partialsublingualglandectomywithexcisionof

theranulahasvalueinclinicalpracticeasanewconservative

method.Nonetheless,thenumberofcasesinthisresearchis

stillsmall,sofurtherinvestigationsareneeded.Moreover,

furtherresearchonfactorssuchastheamountofexcision,

functionalrecovery,andchangeinthepatternofsublingual

glandsafterpartialexcisionisrequired.

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2. BaurmashHD.Acaseagainstsublingualglandremovalasprimarytreatmentofranulas.JOralMaxillofacSurg2007;65:117-21.

3. KurabayashiT,IdaM,YasumotoM,OhbayashiN,YoshinoN,