Central odontogenic fibroma of the maxilla - a case report

Revista

Portuguesa

de

Estomatologia,

Medicina

Dentária

e

Cirurgia

Maxilofacial

ww w . e l s e v i e r . p t / s p e m d

Clinical

case

Central

odontogenic

fibroma

of

the

maxilla

–

A

case

report

Helena

Salgado

,

Pedro

Mesquita

FaculdadedeMedicinaDentáriadaUniversidadedoPorto(FMDUP),Porto,Portugal

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Articlehistory:

Received24October2013 Accepted27January2014 Availableonline4March2014

Keywords: Odontogenictumors Fibroma Adult Humans Pathology Diagnosis

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Central odontogenic fibroma is a benign odontogenic tumor derived from the dental ectomesenchymaltissues.Itisararelesionthatcouldbeseeninallage-groupsandthatit isfoundbothinthemandibleandinthemaxilla.

A24-year-oldcaucasianmalepatientwasreferred,reportingapressureontheleftside oftheupperjaw.Therewerenoothersymptomsandnorecenthistoryofpain.Theclinical examinationdidnotrevealbuccalorpalatalexpansion.Theleftmaxillarysecondpremolar wasmobile.Radiographicevaluationshowedthepresenceofaunilocularradiolucent well-definedareaaroundtherootsofthemaxillaryleftcanine,firstandsecondpremolars.Root canaltreatmentwascarriedoutinteeth23,24and25.Thelesionwassurgicallyremoved undergeneralanesthesia.Thehistopathologicaldiagnosisconfirmedcentralodontogenic fibroma.Thepatientshowednoclinicalorradiographicsignsofrecurrenceoneyearafter surgicalexcision.

©2013SociedadePortuguesadeEstomatologiaeMedicinaDentária.Publishedby ElsevierEspaña,S.L.Allrightsreserved.

Fibroma

Odontogénico

Central

da

Maxila

–

Caso

Clínico

Palavras-chave: Tumoresodontogénicos Fibroma Adultos Humanos Patologia Diagnostico

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O fibroma odontogénico central é um tumor odontogénico benigno que deriva do ectomesênquimadentário.Éumapatologiararaqueatingeváriasfaixasetáriasequetanto podeafetaramandíbulacomoamaxila.

Umindivíduode24 anos,rac¸acaucasina,veioàconsultademedicinadentária apre-sentandoumasensac¸ãodepressãonoladoesquerdodamaxila.Paraalémdesteaspetoo pacientenãoreferiamaissintomatologia.Aoexameclínicofoipossívelverificaraausência deabaulamentodascorticaisósseas.Odente25apresentavamobilidade.Apósrealizac¸ão deexameradiográficoverificou-seapresenc¸adeumalesãounilocularradiotranslúcida, comcontornosbemdefinidos,naproximidadedasraízesdosdentes23,24e25.Efetuou-se aexéresecirúrgicadalesão,soboefeitodeanestesiageral.Oexameanatomo-patológico confirmouodiagnósticodefibromaodontogénicocentral.Opacientenãoapresentasinais clínicosouradiográficosderecidivaapósumanodaremoc¸ãodalesão.

©2013SociedadePortuguesadeEstomatologiaeMedicinaDentária.Publicadopor ElsevierEspaña,S.L.Todososdireitosreservados. ∗ _{Correspondingauthor.}

E-mailaddress:helenatsalgado@gmail.com(H.Salgado).

1646-2890/$–seefrontmatter©2013SociedadePortuguesadeEstomatologiaeMedicinaDentária.PublishedbyElsevierEspaña,S.L.Allrightsreserved.

50

Introduction

Centralodontogenicfibroma(COF)isanextremelyrarebenign tumoraccountingforonly0.1%ofallodontogenictumors.1

Thislesionisconsideredtobederivedfromectomesenchymal tissueofdentaloriginsuchasperiodontalligament,dental papilla,ordentalfollicle.2_{Histologically,thislesionis}

char-acterizedbythe presenceofcolagenousfibrous connective tissue containing varying amounts of odontogenic epithe-lium.Clinically,it presentsasaslowgrowthasymptomatic masswhich,inmostcases,canremainunknownuntilthe appearanceofaswelling.Inmoreseverecasesroot resorp-tionanddisplacementofadjacentteethhavebeenreported.3

COFappearsinboththemandibleandmaxilla(55%and45% respectively).Inthemaxillaithasatendencytoinvolvethe anteriorareawhereasinthemandiblethemolarand premo-larareasarethemostprevalentsites.2,4_{Itisreportedtooccur}

inawideagegroupwithafemalepredilection.5,6_MostCOFs

presentasaradiolucentunilocularlesionwithwell-defined contoursthatcansimulatelesionsofendodonticorigin. How-ever they may also appear as multilocular lesions and in rareinstances mayexhibitamixedradiolucent/radiopaque appearance with poorly defined or diffused borders.2

Enu-cleationorsurgicalcurettageistheappropriatetherapyand recurrenceislow.7

Fig.1–Preoperativepanoramicradiography.

Case

report

A 24-year-old caucasian male patient attended our dental medicine appointmentreferringapressure ontheleft side of the maxilla. Besides this the patient reported no other symptoms. On theclinical examinationwe foundtooth 25 withmobility.Apanoramicradiograph(Fig.1)wasobtained,

Fig.3–Computerizedaxialtomography–frontalslices.

whichrevealedthepresenceofalargeradiolucentlesionnear theroots ofteeth23,24and 25.Thepatient wasasked to takeacomputerizedaxialtomographyinordertoassessthe size of the lesion and its relationship with the surround-ing anatomical structures (Figs. 2and 3). Thetomography showedaverylargelesionthathasalreadyinducedagreat reabsorptionofthelabial plate andthe floorofthe maxil-larysinus.Itwasproceededtherootcanaltreatmentofthe referredteethandthelesionwassurgicallyremoved.Under generalanesthesia,anexcisional biopsy was performed. A bucalmucoperiostealflapwasraisedandthelesionandthe corticalbonethatsurroundeditwereexposed.After remov-ingthecorticalplate,thesoftlesionwasenucleatedfromthe bone.Thesurgicalspecimenwasfixedin10%neutral forma-linandsubmittedtohistopathologicalexamination(Figs.4–6). Grossexaminationrevealedafriablerussetmass,measuring 2.1cm×1.9cm×0.8cm. The histopathological examination (Fig.7)revealedatumorcomposedofacollagenousstroma richinspindleshapedfibroblastswithoutcellularatypia.Few nestsofodontogenicepitheliumwerefound.Nohardtissue suchasboneorcementum-likematerialwasobserved.Alow mitoticactivityandmononuclearinflammatorycellscouldbe found.Animmunocytochemicalanalysiswasperformed to searchthepresenceofvimentin(Fig.8).Theexpressionofthis proteinwasobservedinspindlecells.Correlationofclinical, radiographicandhistopathologicalfeaturesleadtoadefinitive

diagnosis of central odontogenic fibroma, epithelium-poor type. Eighteenmonths aftersurgerya radiographiccontrol (Fig. 9) was made and it was found that trabecular bone becamedenserwhichdemonstratesthatnormalregeneration ofthebonelostbythepressureexertedbythefibromawas occurring.

Fig.4–Photographsofsurgicalremovaloffibroma–buccal

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Fig.5–Photographsofsurgicalremovaloffibroma–lesion exposed.

Fig.6–Photographsofsurgicalremovaloffibroma–after lesionenucleation.

Fig.7–Histologicfeatureofbiopsyspecimen (hematoxylin–eosinstain).

Fig.8–Histologicfeatureofbiopsyspecimen

(immunocytochemicalanalysis–vimentin).

Fig.9–Controlpanoramicradiograph18monthsafter

removalofthelesion.

Discussion

and

conclusions

Central Odontogenic Fibroma isdescribed inthe literature asabenignneoplasmusuallydiagnosedonthesecondand thirddecadeoflifewhichisinaccordancewithourpatient.8,9

Mostauthorsdescribethislesionasbeingmorecommonin females8,10_{despiteinourcaseithasoccurredinamale.}1,10

ThismatchesupwiththestudiesofBuchneretal.11_In2004,

Daniels et al.,1 _{in areviewof the literature,shows a ratio}

ofincidenceonthemandibleandmaxillaof1:1witha70% female-occurrence(49of70).Onthemandible26of35cases occurredmainlyattheposteriorregion,andonthemaxilla26 outof35ofthecasesoccurredattheanteriorregion.Itwas foundtooccurbetweentheages4and80years.Ona retro-spectivestudyof8clinicalcasesofCOF,Hrichietal.12_founda

predilectionformalesex(1.67:1)andthemostcommon loca-tionofthetumorwasonthemandible.Theaverageagewas 19.9yearswithanagerangeof11–38years.Ourcaseisvery similartothatdescribedbySakamotoetal.13_{concerningthe}

ageandsexofthepatientandthelocationofthelesion(near teeth23,24and25).

AccordingtoGardner,14_{therearetwotypesofCOF:}

Fibrousneoplasmorsimpletypewithcollagenousfibrous connectivetissuecontainingodontogenicepithelium; WHOtypewhichapartfromtheabovefeatures,alsoshows presenceofdysplasticdentinorcementumliketissue;

Radiologically,themajorityofcentralodontogenic fibro-mas are radiolucent. It is originally thought that most of theselesionshavemultilocularradiolucencies,butthecurrent reportsshowthattherearemoreunilocularthan multiloc-ular radiolucencies. Thesmall lesions are unilocular while thelargeronestendtobemultilocularortohavescalloped margins.Insomecases, duetothelocation nearthe teeth roots, this tumor can mimic anendodontic origin lesion.15

Periapicalradiolucenciesofnonendodonticoriginoccur infre-quently. When they dooccur, however, a proper diagnosis mustbemade.Dentalprofessionalsmustnotbecome com-placent on the diagnosis of periapical pathoses, but must beawareofotheretiologies.TheCOFisonesuchexample. Althoughithasbeenrarelyreported, itmustbeconsidered ondifferentialdiagnosis foraradiolucencyassociatedwith therootsofvitalandnonvitalteeth.Covanietal.2_presented

acaseofCOFassociatedwiththerootofaneruptedtooth. Theauthorsarguethe importanceofmaking acorrect dif-ferentialdiagnosis ofCOFwithendodontic lesionsshowing thesameradiologicallucentimage.ThediagnosisoftheCOF is determined by its histology. In our case the lesion was associatedwiththerootsofthreeteethandtheprovisional diagnosishadbeenalesionofendodonticorigin.Aftersurgical removalthehistologicexaminationrevealedthereal diagno-sis–COF.

COFisbelieved to arisefrom the odontogenic ectomes-enchyme.Beingamixedtumor,thislesioncanbeconfused withdesmoplasticfibroma(absenceofodontogenicepithelial rests),ameloblasticfibroma(ifodontogenicislandsarelarge andnumerous)ormyxofibromas(collagenfibersalongwith stellate/spindlecells).4,16 _{COFradiographicpresentationcan}

beverysimilartoadentigerouscyst-likelesion,which sug-geststhatCOFshouldbeincludedinthedifferentialdiagnosis ofpericoronallesionsontheposteriormandible.1

As COF is considered a benign odontogenic tumor the treatment of choice is enucleation with careful follow up forafewyears,althoughfewcasesofrecurrencehavebeen reported.17–19 _{Dunlap and Barker}20 _{presentedtwo cases of}

maxillaryodontogenic fibroma treated bycurettage with a follow-upof9and10yearswithnoevidenceofrecurrence. Alsononeofthe8casesreportedbyHrichietal.12_showed

recurrenceonafollowupof2yearsaftersurgery.

Despitebeinganextremelyraretumoritisveryimportant thatdentistsbeawareofitsclinical,radiographicand histolog-icalfeaturesinordertoincludeitonthedifferentialdiagnosis ofodontogenictumors.

Ethical

disclosures

Protection of human and animal subjects.The authors

declarethatnoexperimentswereperformedonhumansor animalsforthisinvestigation.

Confidentiality of data.Theauthors declarethat theyhave followed theprotocols oftheir workcenter onthe publica-tionofpatientdataandthatallthepatientsincludedinthe study have received sufficientinformation and have given theirinformedconsentinwritingtoparticipateinthatstudy.

Right to privacy and informed consent.The authors must

haveobtainedtheinformed consentofthe patientsand/or subjectsmentionedinthearticle.Theauthorfor correspon-dencemustbeinpossessionofthisdocument.

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