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Calcifying epithelial odontogenic tumor associated with the left mandibular first premolar: a case report and literature review

Won-Ki Kim1, Min-Soo Kim1, Eui-Mook Lee1, Jae-Won Cha1, Bo-Young Choi1,

Bong-Chul Kim1, Seung-Ki Min1,2, Jun Lee1,2

1Department of Oral and Maxillofacial Surgery, Daejeon Dental Hospital, School of Dentistry, Wonkwang University, 2Wonkwang Bone Regeneration Research Institute, Daejeon, Korea

Abstract (J Korean Assoc Oral Maxillofac Surg 2012;38:166-70)

Calcifying epithelial odontogenic tumor (CEOT) is a rarely reported benign tumor, accounting for 0.4-3% of all odontogenic tumors. Approximately 150 cases have been reported in the literature between 1958 and 2003. The age range of CEOT varies from 8 to 92 years with mean of 36.9 years, and the occurrence of the lesion in both genders is almost equal. It has 2 clinico-topographic variants: the intraosseous (94%) and the extraosseous (6%) type. The intraosseous type has a predilection for mandible (maxilla : mandible ratio of 1 : 2). The intraosseous CEOT commonly associated with non-erupted teeth accounts for more than half (52%) of the cases and usually appears as painless swelling that causes bony expansion. The location of diffused round-shaped calcifying material is inside the connective tissue stroma and epithelial islands. The tumors tend to be located toward the tooth crown, which usually has a unilocular radiolucent region containing variant radiopaque materials radiologically. In this paper, we report a case of CEOT occurring in the left mandibular first premolar of a 23-year-old female and present a brief review of the literature.

Key word: Calcifying epithelial odontogenic tumor[paper submitted 2011. 8. 7 / revised 2011. 10. 13 / accepted 2011. 10. 15]

timeshigherthanthatofthepremolarregion.About52%of

intraosseousCEOTusuallyappearsinassociationwithnon-

eruptedorimpactedteethandasapainlessswellingmass

withcorticalboneexpansion3.Ithasbiologicalproperties

similartothoseofintraosseousameloblastomaandnormally

occurs in the reducedenamel epitheliumon thedental

laminaremnantsand toothcrown.Asfor thedifferences

betweenCEOTandameloblastoma,theepithelialcellsdo

notlooklikeameloblast,andthelocationofdiffusedround-

shapedcalcifyingmaterial is inside theconnective tissue

stromaandepithelialislands4.Thetumorstendtobelocated

towardthecrownofnon-eruptedteeth,whichusuallyhasa

unilocularradiolucentregioncontainingvariantradiopaque

materialsradiologically.CEOTcontainscalcifyingmassesor

homogenousnon-cellularmaterialwithinthetumorepithe-

liumandstroma5.Inthispaper,wereportacaseofCEOT

occurringintheleftmandibularfirstpremolarofa23-year-

oldfemalepatientandpresentabriefreviewoftheliterature.

II. Case Report

A23-year-old femalepatientvisitedourhospitalwith

I. Introduction

Calcifyingepithelialodontogenictumor(CEOT)isknown

asapindborgtumorandisararelyreportedtumoraccounting

for0.4-3%ofallodontogenictumors1.Approximately150

caseshavebeenreportedbetween1958and2003,butthere

is insufficientevidencetoindicatethelong-termbehavior

ofthistumor2.TheagerangeofCEOTvariesfrom8to92

yearswithmeanof36.9years,andtheoccurrenceof the

lesioninbothgendersisknowntobealmostequal2,3.It is

largelyclassifiedasintraosseous(94%,central)andextra-

osseoustype(6%,peripheral).Theintraosseoustypehasa

predilectionformandible(maxilla:mandibleratioof1:2),

with theprevalence rateof themolar regionabout three

Jun LeeDepartment of Oral and Maxillofacial Surgery, Daejeon Dental Hospital, School of Dentistry, Wonkwang University, 77 Dunsan-ro, Seo-gu, Daejeon 302-830, KoreaTEL: +82-42-341-2800 FAX: +82-42-366-1115E-mail: omslee@daum.net

*ThispaperwassupportedbyaresearchgrantfromWonkwangUniversityin2010.

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.2012.38.3.166

pISSN 2234-7550·eISSN 2234-5930

Calcifying epithelial odontogenic tumor associated with the left mandibular first premolar: a case report and literature review

167

lesionssurroundingtheradiopaquelesionsfrom#33to#35

in thepanoramicradiography,butnofistulaformationin

theoralcavitywasobserved.Themandibularcanalpasses

underthebottomofthelesionswithmentalnervethroughthe

buccalcorticalboneofthecrownof#34.(Fig.1)Inthedental

vitalitytestonneighboringteeth,thecoldtestandelectric

pulpaltestalsoshowednegativereaction.Theroutinedental

threedimensionalcomputerized tomographyscans taken

beforethesurgeryshowedexpansionofbuccalcorticalbone

andmandibularcanal,mentalnerveslightlydisplacedtothe

bottomin#34andclearradiopaquelesionsintheradiolucent

lesionswithobviousboundary.

Reconstructivesurgeryonbonedefectswasperformed

undergeneralanesthesiathroughplannedmassremovaland

tibialbonegraftingonSeptember16,2010.Suchoperation

includedsulcularincision,flapelevation,detachingthemass

fromtheneighboringtissues,extractionoftherelatedtooth

origin(#34),andmassremoval.Therewerenofindingsonthe

chiefcomplaintofdiscomfortfromtheleftmaxillarythird

molar(#28)andfeelingofirritationwhenswallowing.The

panoramicradiographyshowedaradiolucent imagewith

clearboundaryofradiopaquelesionsaroundtheimpacted

teethandcrownoftheleftmandibularfirstpremolar(#34).

Noremarkablesymptoms-suchaspain,edema,bleeding,

androot resorptionofneighboring teeth(leftmandibular

canine, leftmandibularsecondpremolar)-wereobserved,

andtherootsofneighboringteethwerediverged.Withno

mobilityandreactiontothepercussionofneighboringteeth,

therewerenosensationofpainfulpressure,indurationand

fluctuationuponpalpationintherelatedarea.

Shehasnomedicalhistoryofsurgeryrelatedtotheleft

mandibularpremolarorsignificantsystemictreatmentrecord

excepttakingpreventivemedicinefortheslightincreaseof

thethyroidhormonelevelandtakingmedicineforchronic

rhinitis.

Whenshevisitedourhospital, therewere radiolucent

Fig. 1. The radiolucent mass contains some agg lomerated rad iopaque materials around the impacted left mandibular f i rst premolar (A) and Dental CT view (sagittal view) (B). The mandibular canal passes just beneath the mass.Won-Ki Kim et al: Calcifying epithelial odontogenic tumor associated with the left mandibular first pre-molar: a case report and literature review. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 2. The lesion does not appear as bony perforation but slightly bony expansion (A) , and the crown is exposed (B). Calcifying materials are scattered on the inner side of mass (C) and after tibial bone graft (D).Won-Ki Kim et al: Calcifying epithelial odontogenic tumor associated with the left mandibular first pre-molar: a case report and literature review. J Korean Assoc Oral Maxillofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:166-70

168

logicaldevelopmenthasyet toberevealedclearly, there

aretheoriesthatitoriginatedwiththestratumintermedium

ofdentalorgan6andwithdental lamina7.CEOToccursin

allagerangesbutmainlyinthethirdtofifthdecadesinage

groups,withnospecialdifferencebygender8.Themain

commonlocationis theposteriorregionofmandible,and

it isaclinicallypainlessgrowingmassmainly.Incaseof

occurrence in theupper jaw,however, itbehavesmore

aggressivelytoincludenasalcavityormaxillarysinus,and

itcanbeaccompaniedbyclinicalsymptomssuchasnasal

bleeding,nasalobstruction,andheadache.Radiologically,

itcanbeshownasunilocularormultilocular radiolucent

imagewithclearboundary,containingradiopaquematerials

ofvarioussizesanddensitiesinmanycases.Inabout50%

ofthetotalcasesofoccurrence,itappearsinconnectionwith

theimpactedteethorcrownofnon-eruptedteeth9.Incases

whereinnoradiopaquematerialsare included, itmaybe

mistakenasadentigerouscyst.CEOThaslowertendency

topenetrateintothebonytrabecularspacecomparedwith

ameloblastomawhich ismore invasiveform9.Malignant

changeisalsoextremelyrare5.

Histologically, it isarrangedintheformofislandswith

polygonalepithelial cells falling, connected strands,or

thinsheets itssurroundingsarefibrousstromacomposed

ofatypicalacidophilicamyloidtissueandcalcifiedtissue.

(Fig.4)There is an intercellularbridgeconnecting the

polygonalcells.Homogeneousacidophilicmaterialsare

observedbetweenepithelialcells, andvarious formsof

calcifyingmaterialsarefoundmainlyinitssurroundings.The

histologicalclassificationcaninclude(1)thecasewithsmall

amyloidandcalcifyingtissues,(2)thecasewithfullamyloid

andcalcifying tissues,and (3) theclearcellvariant, the

dominantclearcellasepithelialcellwithclearcytoplasm4,9-12.

CEOT’s twohistologicalcharacteristics-calcifyingand

amyloidformation-areconsidereddestructiveyetproductive

forself-limitedgrowth9.Thisacidophiliccalcificationisstill

controversial,buttherearemanyopinionsononeamyloid

typeduetoitspositivereactiontoCongoredorthioflavineT.

However,therearealsoopinionsonthekindofdegenerated

tissue,i.e.,typeIVcollagenorbasallamina,enamel,ordead

skincell13.

Inthiscase,theclearcellwasnotobservedhistologically

it isregardedasthecasewithfullamyloidandcalcifying

tissuesbasedontheclassificationabove.Iftheclearcellis

predominant, it iscalledaclearcellvariant,a typethat is

wellobservedintheperipherallesion(extraosseoustype)4.In

addition,thedestructionofcorticalboneasanintraosseous

massattachmentwiththeneighboringtissues,andthenormal

neighboringtissueswereproperlypreserved.Toreconstruct

thedefectiveregion,particulatedmarrowandcancellousbone

(PMCB)measuring2×2cmwascollectedfromthelefttibia.

AfterthebonedefectswerefilledwithPMCB,absorbable

atelo-collagensponge(Teruplug;TermoCo.,Tokyo,Japan)

wasusedasbarriermembraneonthebuccalside.(Fig.2)

BiopsiesbyHematoxylinandeosinstaining,Congored

staining,andpolarizingmicroscopewereperformedforthe

mass.Avarietyofacidophilicamyloidtissuesandcalcifying

tissuescouldbeobservedfromsuchbiopsies,includingthe

clusterofpolygonalepithelial tissuestrands.Theamyloid

tissuescanbeseenwithredcolorinCongoredstainingand

withbrightgreencolorinpolarizingmicroscope,whichis

usedforanalyzingamyloidmaterialbypolarizationofdouble

retractionmethod.Sincethesurgeryresultwasfavorable,she

wasdischargedfromthehospitalfourdaysafterthesurgery,

andthestitchoutwasperformedintheoutpatientclinic.The

panoramicradiographytakenafterthesurgeryonSeptember

18,2010showedgoodhealingconditionaftermassremoval

andtibialbonegraftingsurgery.(Fig.3)Sensoryparalysisin

theleftlowerlippersisted,butsuchwasprobablyduetothe

slighttensionappliedtothementalnerveintheretraction

processduring thesurgery.Theextractionprocedurefor

tooth#28asthepatient’schiefcomplaintwasperformed.

Observationsincludingthepossibilityoftumorrecurrence

areunderwayonathree-monthbasisintheoutpatientclinic,

and therearenospecificcomplicationsand findingsof

recurrencetodate.

III. Discussion

CEOTisa relatively rarely reported tumoraccounting

forabout1%ofallodontogenictumors3,4.Sinceitshisto-

Fig. 3. Post-operative panoramic radiography.Won-Ki Kim et al: Calcifying epithelial odontogenic tumor associated with the left man-dibular first premolar: a case report and literature review. J Korean Assoc Oral Maxil-lofac Surg 2012

Calcifying epithelial odontogenic tumor associated with the left mandibular first premolar: a case report and literature review

169

uniloculartype,withthelesionssurroundedbyclearradio-

paqueborders.Thecomputedtomographyscansalsoshowed

slightexpansionofcorticalbonewithoutanydestructionof

thebone.Massremovalandbordergrindingprocedureswere

lesionwasnotobservedinthiscase, throughwhichitcan

beinferredasa typewithlessaggression.However,each

classificationhassimilarrecurrencerate.

ThemixedtumorofCEOTandadenomatoidodontogenic

tumorAOTwasfirst reportedbyDammetal.14 in1983.

Inaddition,Chengetal.1presentedareportonthegenuine

malignantCEOTin2002.Thecasepresentedfor thefirst

timein thispaperrevealedhistologicallystrongstaining,

abnormalcelldivision,andpolymorphismtogetherwith

swellingintherightmandibularbody1.

ThetreatmentsforCEOTaregenerallydiverse,ranging

fromenucleationandcurettage tomandibulectomyand

maxillectomyas themore fundamentalprocedures3.The

choiceoftreatmentwilldifferdependingontheradiological,

histological,andclinicalfindingsoneachlesion.

Thepresentcaseshowedself-limitedgrowthitwasofthe

Fig. 4. Deep-stained calcifying materials and eosinophilic amyloids. The epithelial cells shaped like strands surround the calcifying materials. The amyloids show red color in Congo red staining and bright apple green color- which means bi-refringence-in polarizing microscopic view. A. Optical microscopic view (H&E staining, ×40). B. Optical microscopic view (H&E staining, ×200). C. Optical microscopic view (Congo red staining, ×200). D. Polarizing microscopic view (Congo red staining, ×200).Won-Ki Kim et al: Calcifying epithelial odontogenic tumor associated with the left mandibular first premolar: a case report and literature review. J Korean Assoc Oral Maxillofac Surg 2012

Fig. 5. Post-operative 10-month panoramic radiography.Won-Ki Kim et al: Calcifying epithelial odontogenic tumor associated with the left man-dibular first premolar: a case report and literature review. J Korean Assoc Oral Maxil-lofac Surg 2012

J Korean Assoc Oral Maxillofac Surg 2012;38:166-70

170

3. PhilipsenHP,ReichartPA.Calcifyingepithelialodontogenictumour:biologicalprofilebasedon181casesfromtheliterature.OralOncol2000;36:17-26.

4. NevilleBW,DammDD,AllenCM,BouquotJE.Oralandmaxillo-facialpathology.3rded.Philadelphia:W.B.Saunders;2002.

5. DeboniMC,Naclério-HomemMdaG,PintoJuniorDS,TrainaAA,CavalcantiMG.Clinical,radiologicalandhistologicalfeaturesofcalcifyingepithelialodontogenictumor:casereport.BrazDentJ2006;17:171-4.

6. GonF.Thecalcifyingepithelialodontogenictumor:reportofacaseandastudyofitshistogenesis.BrJCancer1965;19:39-50.

7. TakedaY,SuzukiA,SekiyamaS.Peripheralcalcifyingepithelialodontogenictumor.OralSurgOralMedOralPathol1983;56:71-5.

8. GopalakrishnanR,SimontonS,RohrerMD,KoutlasIG.Cysticvariantofcalcifyingepithelialodontogenictumor.OralSurgOralMedOralPatholOralRadiolEndod2006;102:773-7.

9. FranklinCD,PindborgJJ.Thecalcifyingepithelialodontogenictumor.Areviewandanalysisof113cases.OralSurgOralMedOralPathol1976;42:753-65.

10. AnaviY,Kaplan I,CitirM,CalderonS. lear-cellvariantofcalcifyingepithelialodontogenictumor:clinicalandradiographiccharacteristics.OralSurgOralMedOralPatholOralRadiolEndod2003;95:332-9.

11. KumamotoH,SatoI,TatenoH,YokoyamaJ,TakahashiT,OoyaK.Clearcellvariantofcalcifyingepithelialodontogenictumor(CEOT)inthemaxilla:reportofacasewithimmunohistochemicalandultrastructuralinvestigations.JOralPatholMed1999;28:187-91.

12. Schmidt-WesthausenA,PhilipsenHP,ReichartPA.Clearcellcalcifyingepithelialodontogenictumor.Acasereport.IntJOralMaxillofacSurg1992;21:47-9.

13. SappJP,EversoleLR,WysockiGP.Contemporaryoral andmaxillofacialpathology.2nded.NewYork:Elsevier;2004.

14. DammDD,WhiteDK,DrummondJF,PoindexterJB,HenryBB.Combinedepithelialodontogenictumor:adenomatoidodontogenictumorandcalcifyingepithelialodontogenictumor.OralSurgOralMedOralPathol1983;55:487-96.

carriedoutaccordingly,with thepanoramicradiography

taken10monthsafter thesurgeryshowingnofindingsof

recurrence.(Fig.5)

Giventheshortfollow-upperiod, itseemsprematureto

makeaconclusionon the long-termprognosis from the

presentcase.Nonetheless,CEOTisgenerallydefinedasan

aggressivetumorwithabout14%recurrencerate9. In this

case,itiscomparedwithinvasiveameloblastoma,andamore

invasive,aggressivetreatmentplanmayberecommended8.

However,recentstudiesreferredtoCEOTaslessaggressive

tumor3,4andrecommendedconservativeenucleationandthin

bordergrindingintheneighboringboneastreatmentplan4.

Insummary,wepresentedareporttogetherwithareview

ofrelatedliteratureonacaseshowinggoodprogressafter

massremovalandtibialbonegraftingprocedureinCEOT,an

extremelyraretypeamongtheodontogenicbenigntumors.

More long-termfollow-up isbelieved tobenecessary to

monitorfuturerecurrenceandstabilityafterthesurgery.

References

1. ChengYS,Wright JM,WalstadWR,FinnMD.Calcifyingepithelialodontogenic tumorshowingmicroscopic featuresofpotentialmalignantbehavior.OralSurgOralMedOralPatholOralRadiolEndod2002;93:287-95.

2. RapidisAD,StavrianosSD,AndressakisD,LagogiannisG,BertinPM.Calcifyingepithelialodontogenic tumor (CEOT)of themandible:clinicaltherapeuticconference.JOralMaxillofacSurg2005;63:1337-47.