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Warthin’s tumor of the parotid gland: a case report

Chul-Hwan Kim, Sung-Il Han, Moon-Young Kim

Department of Oral and Maxillofacial Surgery, College of Dentistry, Dankook University, Cheonan, Korea

Abstract (J Korean Assoc Oral Maxillofac Surg 2012;38:366-70)

Initially described in 1929, Warthin’s tumor is a benign neoplasm predominantly found in the parotid gland of the salivary glands. Warthin’s tumor is synonymous for cystadenoma lymphomatosum, adenolymphoma, and cystadenolymphoma. This tumor usually causes minimal pain, but the patient may complain of tinnitus, pain in the ear, or deafness. The tumor is treated with surgical excision, which is easily performed due to the superficial location of the tumor. In our case, a 69-year-old man visited our clinic primarily complaining of swelling in the left parotid gland area. Computed tomography revealed well-defined margins of the tumor in the superficial lobe of the left parotid gland and heterogeneous contrast enhancement. We performed superficial parotidectomy, with the pathological examination confirming Warthin’s tumor. We report a typical case of Warthin’s tumor and a literature review.

Key words: Salivary gland, Neoplasms, Adenolymphoma, Oral surgery [paper submitted 2012. 3. 2 / revised 1st 2012. 5. 22, 2nd 2012. 6. 12 / accepted 2012. 6. 13]

fromitthanwomenataratioof5:12,3.Unlikebenignmixed

tumors,itrecursinlessthan2%,andonly1%developsinto

malignanttumor.Therefore,thetreatmentprincipleisthatit

istrackedafterconservativeexcision4.

ThereareseveralhypothesesconcerningWarthin’stumor,

butthewidelyacceptedtheoryexplainsitasaconditionthat

developsfromthehyperplasiaofsalivaryductcellsisolated

inthelymphnodeoftheparotidglandduringembryogenesis.

Other theoriessuggest thatWarthin’s tumor iscausedby

theinfiltrationoflymphocyteinsidetheexistingadenoma

or interactionofhyperplasiaofgrandularepitheliumand

excessivelymphaltissuesinsidethestroma5,6.

Thiscase reportconcerns thesuccessfulexcisionofa

typicalcaseofWarthin’stumorfoundintheparotidgland,

withitsclinicalandradiologicalfeaturesandsurgicalpro-

cedurespresentedincludingstudiesinthereferences.

II. Case Report

A69-year-oldmalepatientvisitedthedentalclinicattached

toDankookUniversity,complainingofswellingonhisleft

cheek.Hehadbeenawareofthisswellingfor2yearsbuthad

notsoughtmedicalhelpsinceithadnotcausedanypainor

discomfort.Recently,however,hefoundthat theswelling

wasworseningandconsequentlyvisited theclinic.His

medicalhistoryrevealedthathehadbeentreatedwithdrugs

I. Introduction

Asabenignneoplasmofsalivaryglandsandnamedafter

pathologistAldredScottWarthin,Warthin’s tumorhas

synonymssuchasadenolymphoma,cystadenolymphoma,

andpapillarycystadenomalymphomatosum1.Amongthese,

papillarycystadenomalymphomatosumiswidelyrecognized

bypathologistssince it reflects the tumor’scharacteristic

tissueshape.Papillarycystademonareferstothedouble-layer

epitheliumwithpapillarybumpsinsidethecystoma,whereas

lymphomatosumpertains to the lymphal tissuessurroun-

dingandsupportingthetumor.Warthinbecamethefirstto

substantiate this tumor,whichhasbeenwidelyknownas

Warthin’stumor-adesignationthatiseasytouse-sincethen2.

This typeof tumor is the secondmost commonafter

pleomorphicadenomaamongotherbenigntumorsfoundin

thesalivaryglands.Itismostfrequentlyfoundintheparotid

glandamongmiddle-agedpatients,withmoremensuffering

Chul-Hwan KimDepartment of Oral and Maxillofacial Surgery, College of Dentistry, Dankook University, 119, Dandae-ro, Dongnam-gu, Cheonan 330-714, KoreaTEL: +82-41-550-1991 FAX: +82-41-551-8988E-mail: kimchoms@dankook.ac.kr

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.2012.38.6.366

pISSN 2234-7550·eISSN 2234-5930

Warthin's tumor of the parotid gland: a case report

367

Theskin incisionuponsurgerystartedfromthe tragus

andextendedbackwardalong thecurveof theear to the

bottomofthemandibularangieasavariantBlairincision.A

vasoconstrictorwasinjected,withskinincisionandplatysma

incisiondonefollowedbytissuedissection toexpose the

investinglayeroftheparotidglandandwell-encapsulated

tumor.Themaintrunkwasfirst identified,andthebranch

wasdissected;thetumorandtheinvestinglayeroftheparotid

glandwerethencarefullyremoved.(Fig.3)Duetotimeand

costconstraints,noothertestwasconductedasidefromCT;

thus,toruleoutthepossibilityofamalignanttumor,some

tissueswereremovedandsubjectedtoafrozensectiontest.

Theresultshowedthatthetumorwasbenign,soadrainage

tubewasinsertedandsuturingwasdonebylayer.(Fig.4)The

excisedtumormeasured3.5×3.5cmandconsistedofgrayish,

yellowish,andgelatinousmaterial.(Figs.5,6)

Aftersurgery,thepatientdidnotexperienceanyparticular

symptomsuchasfacialnervepalsy;hewasdischargedin6

daysaftertheremovalofsuturematerial.

forhypertensionfor20yearsand thatheused tosmoke

halfapackofcigarettesadayfor30yearsbuthadgiven

upsmoking2yearsbefore.Ahard, immobile tumorwith

diameterof4cmwasfoundaroundhisleftmandibularangie

butwithnotenderness,heatsensation,ordifficultyofmouth

openingbypalpationinvolved.Thepatientcomplainedofa

recentamblyacousia.Nointraoralabnormalitywasfound,and

noinfectioninhislymphnodeandotherareaswaspalpated

onhisheadandneck.(Fig.1)

Noabnormalitywasfoundinhispanoramicradiograph.He

wassubjectedtocomputedtomography(CT),whichidentified

awell-defined,contrast-enhancedroundlesionwithdiameter

of3.5×3.5cmandwhichincludedmultiplecystomasand

smallmultiplelymphnodesinLevelsIIandIII.(Fig.2)

Afterconsideringthecombinedclinicalandradiological

testresults,thepatientwasassumedtohaveabenigntumor

likepleomorphicadenomaorWarthin’stumorratherthan

mucoepidermoidcarcinomasincethepatientdidnotfeelpain

andthelesionwaswell-defined.

Fig. 1. Preoperative extraoral findings: tumor of the left parotid gland.Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 2. Preoperative computed tomo-graphy (CT) findings. Enhanced CT image of Warthin’s tumor of the super-ficial lobe of the left parotid gland. 3.5×3.5 cm well-defined margins of the tumor and heterogeneous contrast enhancing with multifocal cystic por-tion. A. Axial view. B. Coronal view.Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:366-70

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parotidglandwasparotidtail,followedbytheanteriormargin

oftheearlobe.Itisattimesfoundinthemouth,mostlyin

thepalateorbuccalmucosa2,9. It isoftenmulticentricand

bilateral(10-30%and5-9%,respectively).Mostcasesare

foundinpopulationaged58-70withrarecases(below6%)

ofbelow40yearsofage10.Fivemenforeverywomanare

knowntosufferfromthiscondition;accordingtoamore

recentlypublishedpaper,however, themaleandfemale

III. Discussion

Asthesecondmostfrequenttypeoftumorfoundinthe

salivarygland,Warthin’stumoraccountsfor14-30%ofall

tumorsfoundintheparotidgland7.Thistumorisat times

foundinthesubmaxillarygland,butismorefrequentlyfound

intheparotidgland,mostlyintheinvestinglayer,butrarely

inthedeeplobe(10%)8.Themostfrequentlocationinthe

Fig. 3. Operative findings. A. Preauricular and retromandibular incision line design (modified Blair incision). B. Superficial paro-tidectomy with Warthin’s tumor. C. The superficial lobe has been removed, and the facial nerve is exposed.Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean As-soc Oral Maxillofac Surg 2012

Fig. 4. Postoperative findings.Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean As-soc Oral Maxillofac Surg 2012

Fig. 5. Gross finding. The cut surface shows a few cystic spaces containing grayish, yellowish, and gelatinous material.Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean As-soc Oral Maxillofac Surg 2012

Warthin's tumor of the parotid gland: a case report

369

spiteofitsexcellentcontrastandeffectivenessinassessing

thelesion.Incontrast,CTisrelativelycheapandquickas

wellasaccurateinidentifyingthelocation,malignancy,and

correlationwithfacialnerves,atthesametimereducingthe

radiationexposure;thusservingasanidealmeansoftesting15.

In recentyears, fineneedleaspirationcytologyhasbeen

widelyusedindiagnosingtheneoplasmofthesalivarygland,

withtestingaccuracyofupto87-97%16.Warthin’stumoris

diagnosablesincetheacidophiltumorcellsandlymphnode

areclearlyvisibleduringsuchatest.Nonetheless,caution

isrequiredsinceupto26%ofWarthin’stumorcasesare

misdiagnosedasmalignant17. In thiscase,CTwas first

performedsinceitwasthesimplestandmostaccuratemeans

ofidentifyingthelesion.Theresultofthetestshowedthatthe

lesionwaslikelybenignsinceitwaswell-defined.Withthe

patientrefusingtobetestedadditionallyduetotime,distance,

andcostconstraints,surgerywasplannedwithoutfurther

tests.

Thereareseveralconflictingopinionsontheidealtreat-

ment ofWarthin’s tumor.Ageneral consensus is that

surgicalremovalisrequired,butthescopeofsurgeryisstill

acontroversialsubject.Batsakis18consideredthis tumor’s

likelihoodofturningmalignanttobeonly0.3%,andthatit

wastheleastharmfultumoramongothertumorsfoundinthe

salivarygland.Thus,theyassertedthatitwascurablewith

enucleation18.Anotherresearchconcludedthatparotidectomy

wasrequiredtoreducetherecurrenceratesinceWarthin’s

tumorislikelytobebilateralandmulticentric7.Sinceit is

largelywell-definedandlocatedonthesurfacelayerofthe

paroticgland,superficialparotidectomyisusuallyused;if

patientratioisnowreducedto1.6:1.Themostconvincing

causeofsuchincreaseamongwomenistheincreaseinthe

numberoffemalesmokers.Accordingtoareport,casesof

Warthin’stumorare8timesmorefrequentamongsmokers

thanamongnon-smokers,withupto90%oftumorpatients

havingahistoryofsmoking;thusshowingclearcorrelation

betweentheoccurrenceof tumorandsmoking11,12. In this

case,too,thepatientwasamiddle-agedmalewhohadalong

historyofsmoking.Thelocationwherethetumorwasfound

wastypical,i.e.,tailpartoftheinvestinglayeroftheparotid

gland.

Warthin’stumorisgenerallyacrystallinelesionfoundas

aroundoranoval-shapedswellingatoraroundtheparotid

gland. Itcauses littleornopainand tinnitus,pain in the

ear,anddeafness insomecases.Thesizecanbeseveral

millimeters to centimeters and2-4 cmon the average.

Accordingtoastudy,56%ofatotalof278subjectshad1-3

cm,and40%,4-6cm,with1subjecthavingover10cm13.In

thiscase,too,thepatientdidnotcomplainofpain,andthe

sizeof thetumorwasaverage.Thepatientcomplainedof

amblyacousia.

Warthin’stumoristestedpriortosurgerybyptyalography,

ultrasonography,magneticresonanceimaging(MRI),and

CT.Ptyalographycanbeusedtoassumeadiagnosiswith“ball

inhandappearance.”Ultrasonographydoesnotexposethe

patienttoradiation;althoughithastheadvantagesofshort

durationandlowercost,ultrasonographycanbeusedonlyas

anauxiliarymeansoftestingsincethetumormaybefound

asasonolucentcysteventhoughitactuallyconsistsofcyst

andsolidtumor14.MRIisexpensive,takingalongtimein

Fig. 6. Microscopic findings. The tumor consists of cystic spaces lined by bilayered oncocytic epithelium and lymphoid stroma (H&E staining, A: x40, B: x200).Chul-Hwan Kim et al: Warthin's tumor of the parotid gland: a case report. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:366-70

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1981;39:362-6.4. HellerKS,AttieJN.TreatmentofWarthin'stumorbyenucleation.

AmJSurg1988;156:294-6.5. AlbrechtH,ArztL.Papillarecystadenomeinlymphdrusen.Frankfurt

ZPathol1910;4:47-69.6. ThompsonAS,BryantHCJr.Histogenesisofpapillarycysta-

denomalymphomatosum(Warthin'stumor)oftheparotidsalivarygland.AmJPathol1950;26:807-49.

7. TeymoortashA,KrasnewiczY,WernerJA.Clinicalfeaturesofcystadenolymphoma(Warthin's tumor)of theparotidgland:aretrospectivecomparativestudyof96cases.OralOncol2006;42:569-73.

8. MaioranoE,LoMuzioL,FaviaG,PiattelliA.Warthin'stumour:astudyof78caseswithemphasisonbilaterality,multifocalityandassociationwithothermalignancies.OralOncol2002;38:35-40.

9. ChapnikJS.ThecontroversyofWarthin'stumor.Laryngoscope1983;93:695-716.

10. YihWY,KratochvilFJ,StewartJC.Intraoralminorsalivaryglandneoplasms:reviewof213cases.JOralMaxillofacSurg2005;63:805-10.

11. LamelasJ,TerryJHJr,AlfonsoAE.Warthin's tumor:multi-centricityandincreasingincidenceinwomen.AmJSurg1987;154:347-51.

12. MonkJSJr,ChurchJS.Warthin'stumor.AhighincidenceandnosexpredominanceincentralPennsylvania.ArchOtolaryngolHeadNeckSurg1992;118:477-8.

13. Párraga-LinaresL,Aguirre-Urízar JM,Berini-AytésL,Gay-EscodaC.Papillarycystoadenomalymphomatosum(Warthin-like)ofminorsalivaryglands.MedOralPatolOralCirBucal2009;14:e597-600.

14. AllegraSR.Warthin's tumor:ahypersensitivitydisease?Ultra-structural,light,andimmunofluorescentstudy.HumPathol1971;2:403-20.

15. KoyuncuM,SeşenT,AkanH,IsmailogluAA,TanyeriY,TekatA,etal.Comparisonofcomputed tomographyandmagneticresonanceimaginginthediagnosisofparotidtumors.OtolaryngolHeadNeckSurg2003;129:726-32.

16. StewartCJ,MacKenzieK,McGarryGW,MowatA.Fine-needleaspirationcytologyofsalivarygland:areviewof341cases.DiagnCytopathol2000;22:139-46.

17. ParwaniAV,AliSZ.Diagnosticaccuracyandpitfalls infine-needle aspiration interpretation ofWarthin tumor.Cancer2003;99:166-71.

18. Batsakis JG.Carcinomaexpapillary cystadenoma lympho-matosum.MalignantWarthin'stumor.AnnOtolRhinolLaryngol1987;96:234-5.

19. MaddoxPT,PaydarfarJA,DaviesL.Parotidectomy:a17-yearinstitutionalexperienceataruralacademicmedicalcenter.AnnOtolRhinolLaryngol2012;121:100-3.

20. O'BrienCJ.Currentmanagementofbenignparotidtumors--theroleoflimitedsuperficialparotidectomy.HeadNeck2003;25:946-52.

thelesionisinthedeeplobe,however,deepparotidectomy

isdoneafterperforming superficialparotidectomyand

confirmingthebranchesoffacialnerves.Note,however,that

parotidectomywithextendedrangereportedlyresults ina

highratioofcomplications.Morespecifically,approximately

8%of thepatientswhoundergosuchsurgeryonthearea

beneath the investing layer reportedFrey’s syndrome19.

Yetanother researchshowed that27%of suchpatients

experiencedfacialnervepalsy,andthat2.5%experienced

permanentfacialnervepalsy20.Therefore, inrecentyears,

partialparotidectomyhasbeenused;itisbasicallythesame

withpalliativesuperficialparotidectomy,but thetumoris

removedtogetherwithnormalparotidglandtissuesinsteadof

removingallinvestinglayersoftheparotidglanddepending

onthelocationofthetumor.SinceWarthin’stumorisoften

foundat the tailpartof theparotidgland,partialparoti-

dectomymaybeanappropriateprocedurebecauseitremoves

onlythebottombranchesofthefacialnervesandthetumor

includingthesurroundingnormaltissues.Inthiscase, the

tumorwaswidelyseatedover the investing layerof the

parotidgland;thus,superficialparotidectomywasconducted

toreducetheriskofrecurrenceinsteadofsimplyremoving

thetumor.

Thiscasewillbepresented togetherwith theclinical

characteristicsand treatmentdetailsandaconsideration

of referencesasanelderlypatientaged69yearshaving

Warthin’stumorthatwasfoundtobetypicalbasedonCTand

yieldingasuccessfulresultfromthesurgerywithnoparticular

complication.

References

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2. ChaudhryAP,GorlinRJ. Papillary cystadenoma lympho-matosum(adenolymphoma);areviewoftheliterature.AmJSurg1958;95:923-31.

3. IbiA,YokobayashiT,KawasakiT,NakajimaT.BilateralWarthin'stumor:reportofcaseandreviewofJapaneseliterature.JOralSurg