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