www.bjorl.org
Brazilian
Journal
of
OTORHINOLARYNGOLOGY
ORIGINAL
ARTICLE
Surgical
treatment
strategy
in
Warthin
tumor
of
the
parotid
gland
夽
Dong
Hoon
Lee
a,b,
Tae
Mi
Yoon
a,b,
Joon
Kyoo
Lee
a,b,∗,
Sang
Chul
Lim
a,baChonnamNationalUniversity,MedicalSchool,DepartmentofOtolaryngology-HeadandNeckSurgery,Hwasun,SouthKorea bChonnamNationalUniversity,HwasunHospital,Hwasun,SouthKorea
Received17November2017;accepted10April2018
Availableonline16May2018
KEYWORDS Warthintumor; Parotidgland; Computed tomography; Fine-needlebiopsy; Surgicalprocedures Abstract
Introduction:Warthintumorsarethesecondmostcommonbenigntumorsoftheparotidgland. WeexaminedtheclinicalfeaturesofWarthintumorsinourhospital,andanalyzedthe consis-tencywithintheliteratures.
Objective:TheaimofthisstudyistoanalyzetheclinicalfeaturesofWarthintumorsinour 10-yearexperienceof118Warthintumorsundergoingsurgeryatasingleinstitute.
Methods:FromDecember2006toDecember2016,110patientswhounderwentsurgical treat-mentforWarthintumorswereidentifiedbasedontheirmedicalrecords.
Results:Atotalof118parotidglandoperationswereperformedin110patients.Almost90% ofWarthintumors werefound inmales,andaveragepatientagewas 66.1±6.1years.The prevalenceofsmokinghistorywas89.1%(98/110).Eightpatients(7.3%)hadbilateralWarthin tumors.Seventy-sevenlesions(65.3%)werelocatedintheparotidtailportion,followedby34 lesionsinthesuperficiallobe(28.8%)and7lesionsinthedeeplobe(5.9%).
Conclusion:Wedetermined theappropriateextentofsurgerydependingonthefineneedle aspiration cytology and tumor location by computed tomography scans. Partial facial dys-function aftertheoperationwas detected in12 cases,andfacialnervefunction recovered within3months.Onlyonepatientexperiencedarecurrence,andwasdiseasefreeafterthe re-operation.Wesuggestthatourtreatmentalgorithm,dependingonthelocationoftumors andtheresultoffineneedleaspirationcytology,canbeusefultodeterminetheappropriate extentofsurgeryforWarthintumors.
© 2018 Associac¸˜ao Brasileira de Otorrinolaringologia e Cirurgia C´ervico-Facial. Published by Elsevier Editora Ltda. This is an open access article under the CC BY license (http:// creativecommons.org/licenses/by/4.0/).
夽 Pleasecitethisarticleas:LeeDH,YoonTM,LeeJK,LimSC.SurgicaltreatmentstrategyinWarthintumoroftheparotidgland.BrazJ Otorhinolaryngol.2019;85:546---50.
∗Correspondingauthor.
E-mail:joonkyoo@jnu.ac.kr(J.K.Lee).
PeerReviewundertheresponsibilityofAssociac¸ãoBrasileiradeOtorrinolaringologiaeCirurgiaCérvico-Facial.
https://doi.org/10.1016/j.bjorl.2018.04.004
1808-8694/©2018Associac¸˜aoBrasileiradeOtorrinolaringologiaeCirurgiaC´ervico-Facial.PublishedbyElsevierEditoraLtda.Thisisanopen
PALAVRAS-CHAVE
TumordeWarthin; Glândulaparótida; Tomografia computadorizada; Biópsiaporagulha fina;
Procedimentos cirúrgicos
EstratégiadetratamentocirúrgiconotumordeWarthindeglândulaparótida
Resumo
Introduc¸ão: OstumoresdeWarthinsãoossegundostumoresbenignosmaiscomunsdaglândula parótida. Avaliamosas características clínicas dos tumores deWarthin em nosso hospitale analisamosaconsistênciacomaliteratura.
Objetivo: AnalisarascaracterísticasclínicasdostumoresdeWarthinemnossaexperiênciade 10anosde118tumoresdeWarthinsubmetidosatratamentocirúrgicoemumúnicoinstituto.
Método: Dedezembrode2006adezembrode2016,110pacientesquereceberamtratamento cirúrgicoparatumoresdeWarthinforamidentificadoscombaseemseusprontuáriosmédicos.
Resultados: Foramfeitas118cirurgiasnaglândulaparótidaem110pacientes.Quase90%dos tumores deWarthin foramencontradosemhomenseamédiadaidadedospacientesfoide 66,1±6,1anos.Aprevalênciadetabagismofoide89,1%(98/110).Oitopacientes(7,3%)tinham tumoresdeWarthinbilateraisnaglândulaparótida.Daslesões,77(65,3%)localizavam-sena porc¸ãodacaudadaparótida,seguidaspor34nolobosuperficial(28,8%)e7noloboprofundo (5,9%).
Conclusão:Determinamosaextensãoapropriadadacirurgiadeacordocomapunc¸ão aspira-tivacomagulhafinaelocalizac¸ãodotumorportomografiacomputadorizada.Disfunc¸ãofacial parcialapósacirurgiafoidetectadaem12casoseafunc¸ãodonervofacialfoirecuperadaem3 meses.Apenasumpacienteapresentourecidivaeficoulivredadoenc¸aapósreoperac¸ão. Suger-imosquenossoalgoritmodetratamento,adependerdalocalizac¸ãodostumoresedoresultado daPAAF,podeserútilparadeterminaraextensãoapropriadadacirurgiaparaostumoresde Warthin.
© 2018 Associac¸˜ao Brasileira de Otorrinolaringologia e Cirurgia C´ervico-Facial. Publicado por Elsevier Editora Ltda. Este ´e um artigo Open Access sob uma licenc¸a CC BY (http:// creativecommons.org/licenses/by/4.0/).
Introduction
Warthintumorsarethesecondmostcommonbenigntumors oftheparotidgland,accountingforabout15%ofallparotid tumors.1---5 Warthin tumorstypically present as an asymp-tomatic, slowly growing mass, usually located in the tail (inferior)portionoftheparotidgland.1,2,5,6Warthintumors occurmorefrequentlyinmales,withapeakincidenceinthe 5thand6thdecades.Thesetumorshaveatendencytoward multiplicity andbilaterality, and have an association with smoking.1---6Recently,manyreportshavepresentedpatients withclinicalfeaturesthatdifferfromthetraditionaltumors insomeway,suchasincreasingincidenceamongfemales, higherratesoftumormultiplicityandbilaterality.4---6In addi-tion, Warthin tumors show regional, national, and racial differences.5
WeexaminedtheclinicalfeaturesofWarthintumorsin patients treatedin our hospital,andanalyzed the consis-tencywithintherelevant literature.The aimofthisstudy wasto analyzethe clinical features of Warthin tumorsin our10-year experienceof 118Warthin tumorsundergoing surgeryatasingleinstitute.
Methods
AfterobtainingapprovalfromtheInstitutionalReviewBoard of ourHospital,a retrospective review (CNUHH-2017-134) wasperformedtoevaluatepatients whoreceivedsurgical
treatment for Warthin tumor of the parotid gland at the Department of Otolaryngology-Head and Neck Surgery in thehospitalfromDecember2006 toDecember2016.One hundredtenpatients whoreceivedsurgical treatment for Warthintumoroftheparotidglandwereidentifiedbasedon theirmedicalrecords.ClinicaldataofpatientswithWarthin tumorof theparotidglandwere reviewed,includingage, sex, underlying diseases, smoking or alcohol, location of tumors,symptoms,durationofsymptoms,fineneedle aspi-rationcytology (FNAC),surgicalprocedures,complications andrecurrence.
Allpatientsunderwentcomputedtomography(CT) scan-ningbeforesurgerytoassesstheextentofthelesionsand toaidin treatment planning.All patients,except forfive patients,underwentFNAC.
The type andextent of surgerywas dependent onthe location of tumors and the result of FNAC. All patients underwentamacroscopicallycompleteoncologicresection. Partialparotidectomy,involving enucleationorremovalof theinferior half ofthe superficial lobe,wasperformed if theWarthintumorwaslocatedinthetail(inferior)portion of the parotid gland. Superficial parotidectomy was per-formedifthetumorwaslocatedinthesuperficiallobe.Total parotidectomywasperformed ifthetumorwaslocatedin thedeeplobe.
Intraoperative facial nerve monitoring was typically used. Postoperative drainage was performed and it was maintainedbyaspiration.AllcasesofWarthintumorswere confirmedhistopathologically.Fisher’sexacttestwasused
for statistical analysis using SPSS version 20.0. Statistical significancewasdefinedasap-value<0.05.
Results
Atotalof118parotidglandoperationswereperformedin 110patients.Clinicaldataof118parotidglandoperations for Warthin tumor aresummarized in Table 1. This group of110 patientsincluded 98 males(89.1%)and 12females (10.9%).Theageatthetimeofdiagnosisrangedbetween37 and85years,withameanof66.1±6.1years.Amongthese 110patients,54 patients(49.1%)hadunderlying diseases, suchashypertension(n=44),diabetes(n=21),chronic hep-atitisB(n=5),andasthma(n=4).Theprevalenceofsmoking historywas 89.1% (98/110), andthe period of pack-years rangedfrom2.4pack-yearsto100pack-yearswithamean periodof 33.4±19.0pack-years.The rateofalcohol con-sumptionwas56.4%(62/110).
Of the 118 lesions, 64 Warthin tumors (54.2%) were locatedin theright parotidgland,and54Warthin tumors (45.8%)werelocatedintheleftparotidgland.Eightpatients (7.3%) had bilateral Warthin tumors. Most of the tumors (107/118, 90.7%) presented as a slowly enlarging mass withintheparotidgland.Theremainingeleventumorswere incidentallydiagnosedbypositronemissiontomography-CT (PET-CT,n=10) or CT (n=1). The majority of the lesions were asymptomatic. The duration of symptoms ranged from0.3months to480months, withamean durationof 20.2±50.6months.ThesizeofWarthintumorsrangedfrom 1cmto7.3cm,withameansizeof2.9±1.0cm.
Amongthe113WarthintumorswhichunderwentFNAC, 68 lesions were diagnosed as Warthin tumors, but the remaining 45 lesions could not be diagnosed as Warthin tumorspreoperatively(Table2).FNAChadadiagnostic sen-sitivityof 60.2%, adiagnostic specificity of 0%,a positive predictivevalueof100%,anegativepredictivevalueof0%, andanaccuracyof60.2%indiagnosingWarthintumors.No specificcomplicationswereobservedafterFNAC.
Seventy-seven lesions (65.3%) were located in the tail portionoftheparotidgland,34lesions(28.8%)werelocated
Table1 Clinicaldataof118parotidglandoperationsfor Warthintumor.
Factors Value
Age(year):mean±SD (range)
66.1±6.1(37---85)
Sex(male/female) 98/12
Smoking(yes/no) 98/12
Periodofpack-years(year): mean±SD(range)
33.4±19.0(2.4---100) Location(right/left/both) 64/54/8
Durationofsymptom (month):mean±SD(range)
20.2±50.6(0.3---480) Sizeoftumor(cm):
mean±SD(range)
2.9±1.0(1---7.3) Periodoffollow-up
(month):mean±SD(range)
50.3±34.1(1---120)
Recurrence 1(0.01%)
SD,standarddeviation.
in the superficial lobe, and 7 lesions(5.9%) were located inthedeeplobe.Amongthe77Warthintumorsinthetail portionof the parotidgland,34 lesionswere removed by superficialparotidectomy,andtheremaining43lesionswere removedbypartialparotidectomy.AllWarthintumorsinthe superficialanddeeplobesof theparotidglandunderwent superficialparotidectomyandtotalparotidectomy, respec-tively(Table3).Themostcommonsurgicalprocedurewas superficialparotidectomy (n=68,57.6%), followedby par-tialparotidectomy(n=43,36.5%)andtotalparotidectomy (n=7,5.9%).
Partial facial dysfunction after the operation was detected in12 cases,andfacialnervefunctionrecovered within3months.Weanalyzedtheriskfactorsthatcancause facialnervedysfunction(Table4).WhenWarthintumorsare locatedinthedeeplobe,theriskoffacialnervedysfunction issignificantlyhigher(p<0.05).Afterthe118surgical proce-dures,5casesdevelopedotherminorcomplications,suchas temporarysalivaryfistula(n=4)orpostoperativehematoma (n=1).Allminorcomplicationsresolveduneventfully.
Themeanfollow-upperiodaftersurgerywas50.3±34.1 months,witharangefrom1to120months.Onlyonepatient experienced arecurrence,and wasdiseasefreeafter the re-operation.
Discussion
PreviousstudieshavesuggestedthatWarthintumorsoccur morecommonlyinmalesolderthan60years.1---6Inthisstudy, almost 90% of Warthin tumors were found in males, and average patient age was 66.1±6.1 years. We also found astrongassociationbetweensmokingandWarthintumors. Theprevalenceofsmokinghistorywasveryhigh(89.1%),as inthepreviousreports.1---6 Eight(7.3%)ofthe110patients hadbilateralWarthintumors.
TheexactpathogenesisofWarthintumorsisunknown.7---9 Thepredominanthypothesissuggeststhatthislesionarises from the salivary gland tissue entrapped within parotid lymphnodes during embryogenesis. Carcinogens in smoke seem tobean importantriskfactor fortheoccurrence of Warthintumors.Severalstudieshavealsoshowntheroleof progesterone receptorsin theetiology ofWarthintumors. There arefew studies assessing the roleof viruses in the pathogenesisofWarthintumor.
Similar to previous reports, an asymptomatic slowly growingmassintheparotidglandwasthepresenting symp-tominthisstudy.1,2,5,6FordiagnosisofWarthintumors,we performedFNACandCTscans.FNACisasimpleand effec-tive method to diagnose a salivary gland tumor.10 In this study,almostallpatientshadundergonepreoperativeFNAC. The sensitivityandspecificityfor thediagnosis ofWarthin tumor were not high. However, all results of FNAC could behelpfultoruleoutmalignancy.Incysticparotidlesions, FNACisdifficultasthesmearsareoftenhypocellularorof poor quality, and donot lead to an accuratediagnosis.11 The reasonfor the lowspecificity maybethe small num-berofsamples.Inthisstudy,FNACresultsofalldeeplobe Warthin tumorswere inaccurate.In small lesionsor deep lobe tumors,ultrasound-guidedFNACmay bemoreuseful thantraditionalblindFNAC.
Table2 Theresultsofpreoperativefine-needleaspirationcytologyassociatedwithtumorlocation. Warthintumorlocation Warthintumor
(n=68)
Benigncysticlesion (n=18) Inflammation (n=16) Sialadenitis (n=11) Tailportion(n=75) 49 12 12 2 Superficiallobe(n=34) 19 6 4 5 Deeplobe(n=4) 4
Table3 Summaryofthesurgicalmethodsassociatedwithtumorlocation. Warthintumorlocation Partial
parotidectomy (n=43) Superficial parotidectomy (n=68) Total parotidectomy (n=7) Tailportion(n=77) 43 34 Superficiallobe(n=34) 34 Deeplobe(n=7) 7
Table4 Analysisofriskfactorsforfacialnervedysfunction.
Facialnervedysfunction Univariateanalysis Multivariate analysis
Yes No
Tumorlocation Deep 3 4 p=0.023 p=0.008
(HR=11.269) Superficialortail 9 102 Tumorsize <2.87cm 3 56 p=0.125 p=0.0058 (HR=0.241) >2.87cm 9 50 HR,hazardratio. Parotid tumor CT,FNAC
Suggestive of Warthin Tumor
No-Diagnosis of Warthin Tumor
Total Parotidectomy Deep Lobe Superficial Lobe Deep Lobe Superficial Lobe
Partial Parotidectomy Superficial Parotidectomy Tail Portion Tail Portion
Diagnosis of Warthin Tumor
Figure1 AlgorithmforthesurgicaltreatmentofWarthintumoroftheparotidgland.
CT provides reliable informationabout the locationof the tumor.5,9,10 In addition, recent developments in CT technology can help identify even the smallest Warthin tumors.10 CT scans of Warthin tumors showed enhanced, well-circumscribedlesionswithcysticcomponentsandsolid stroma.5,9,10 In this study, we could obtain the preopera-tiveinformationandperformtreatment planningby using CTscansandFNAC.
The treatment of choice for Warthin tumors is surgery.5,6,9---12 However, there is a controversy about the
appropriateextent ofsurgery.5,6,12 Inthisstudy,we deter-minedtheappropriateextentofsurgeryforWarthintumors dependingonthelocationoftumorsandtheresultofFNAC (Fig.1).Inourhospital,whenWarthintumorswerefound, surgery was recommended. Periodic CT confirmation was performed on patients who refused surgery. According to thisalgorithm,118parotidglandsurgerieswereperformed without recurrenceexcept in one case. One patient with recurrenceunderwentsuperficialparotidectomyatthe1st surgery. However,the tumor recurredin the same region
9 monthslater, and it wasremoved by partial parotidec-tomy.
Intheliterature,theincidenceoftransientfacialnerve dysfunctionhasbeen reportedtorangefrom8%to68%in allcases.5,6,12 Inthisstudy,theincidenceofpostoperative transientfacialnervedysfunctionwas10.2% (12/118).All casesdeveloped facialnerve dysfunctionafter superficial parotidectomy(n=9)andtotalparotidectomy(n=3).Inthis study,theriskoffacialnervedysfunctionwassignificantly higherinWarthintumorsinthedeeplobethaninsuperficial lobeor tail portion(p<0.05). Other minorcomplications, includingsalivaryfistulaandhematoma,occurredafteronly superficialparotidectomy.Bothfacialdysfunctionandminor complicationsresolveduneventfully.
The limitations of thisstudy arethe smallsample size anda retrospective review. A prospective study including alargesamplesizeisnecessarytoestablishourtreatment algorithmforWarthintumors.
Conclusion
WedemonstratedthatWarthintumorsoccurred more fre-quentlyinelderlymaleswithatendencytowardbilaterality, andanassociationwithsmoking.Ourtreatmentalgorithm, dependingonthelocationoftumorsandtheresultofFNAC, canbeusefultodeterminetheappropriateextentofsurgery forWarthintumors.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
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