Calcifying epithelial odontogenic tumor associated with ... · Calcifying epithelial odontogenic...
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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: [email protected]
*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
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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
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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.