Bilateral osteoid osteoma of the mandible: An unusual case report

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CaseReport

Bilateral osteoid osteoma of the mandible: An unusual case report

T.Adouly*,T. Oubahmane,C. Adnane, S.Rouadi, R.Abada, M.Roubal, M. Mahtar

DepartmentofENT,20AugustHospital,IbnRochdUniversityHospital,Casablanca,Morocco

1. Introduction

In1935,Jaffehasdescribedtheﬁrstcaseofosteoidosteomaand hasconsideredasadistinctclinicalentity[1].Thetruenatureof thislesionisstillunknown.Someauthorsconsideritlikeatrue neoplasmbutothershavereporteditastheconsequenceoftrauma or inﬂammation process [2]. Usually an osteoid osteoma is monofocalandunilocular.Inextremelyrareinstances,itmaybe multifocal(morethanoneosteoidosteomainasinglebone).There areveryfewreportsaboutitsoccurrenceinthecraniofacialbones [3].Wereportanunusualcaseofbilateralosteoidosteomaofthe mandible.Wehighlighttheepidemiological,clinicalaspectsand radiologicalofthisuncommondisease.

2. Casereport

An11-year-oldfemalepresentedtotheENTdepartmentwitha moderatetoseverepain,especiallyatnight,intheleftandright angleofmandiblesince2months.Patientcomplainedofswelling intheleftangleofmandiblewhichhadincreasedinthesizeover thepast2years.Therewasnopriorhistoryoffacialtraumaandno medical history was noted. Physical examination revealed a swelling on the left angle of mandible, measuring about 2cm (Fig. 1). This lesion was rounding, well-deﬁned, hard in

consistency.Itwasimmobileand ﬁxedtotheunderlyingbone.

Theswellingwasnon-erythematous.Therewasnoparesthesiaor anesthesiaoftheface.Intraoralexaminationwasnormal.There were no palpable regional lymph nodes. An axial computed tomography (CT) scan revealed two hypo dense bony masses centered on the cortical bone of the left and right angle of mandible measuring successively (2.48cm) and (0.6cm). The boundaryofthelesionwiththesurroundingscleroticbonewas irregular(Figs.2and3).Undergeneralanesthesia,followingaskin incision and elevation of theperiosteum, thelesion in the left angleofmandibleandtheright’sonewerecompletelyremoved usingcurettageandshaving(Fig.4).Histopathologicalﬁndingsof the two specimens showed proliferation of bone and osteoid sheets,stronglyvascularized,andrichinosteoblasts.Thelesions werediagnosedasosteoidosteoma.Thepostoperativecoursewas uncomplicated. Thespontaneous painimmediatelydisappeared after surgery. The patient was still free of symptoms after 6months.

3. Discussion

Osteoid osteoma is a benign lesion representing 3% of all primarybonetumors,and10%ofbenignbonetumors.About80%

ofosteoidostemaoccurinlongbones,whilelessthan1%occursin jaws[4].InthereviewoftheEnglishliterature,21casesofosteoid ostemas inthejaws werefound.Five osteoidosteomas(23.8%) occurredinthemaxillaand16osteoidosteomas(76.2%)werein themandible[5].

InternationalJournalofPediatricOtorhinolaryngologyExtraxxx(2015)xxx–xxx

ARTICLE INFO

Articlehistory:

Received8March2015

Receivedinrevisedform6April2015 Accepted7April2015

Keywords:

Osteoidosteoma Mandible Pain Multifocal

ABSTRACT

Osteoidosteomaisaslow-growingosteogenictumorwhichhasseldombeendescribedinthejaws.

Usuallyitismonofocalandunilocular.Inextremelyrareinstancesitmaybemultifocal.Thiscasereport highlightstheepidemiological,clinical,andradiologicalfeaturesofthisraredisease.

Wedescribeararecaseofapatientwithbilateralosteoidosteomaofthemandible.Bothofthemwere removedsurgically.Histologicalexaminationconﬁrmeddiagnosis.Thefollow-upforthelast6months wasnormalwithoutrecurrence.

Osteoidosteomaisarareentity.Surgeryisthemainstayoftreatment.Followupisdiscussedinorder todetectanyrecurrence.

ß2015ElsevierIrelandLtd.Allrightsreserved.

* Correspondingauthor.Tel.:+21265826291.

E-mailaddress:adouly.taouﬁk@gmail.com(T.Adouly).

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Pleasecitethisarticleinpressas:T.Adouly,etal.,Bilateralosteoidosteomaofthemandible:Anunusualcasereport,Int.J.Pediatr.

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1871-4048/ß2015ElsevierIrelandLtd.Allrightsreserved.

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This case highlights the seldom multifocal nature of this uncommondisease.Usuallyosteoidosteomaisa solitarylesion, multicentric(involvingmorethanonebone)andmultifocalarea very rare variant [3]. Kyriakos et al. found only 28 patients presentedmultifocalosteoidosteoma;longboneswereinvolved mostfrequentlyandthere wasnojawbone involvement[6]. In 2013,Anetal.presenttheonlycasereportofmultifocalosteoid osteomawith4nidiinvolvingthemandible[5],whichexplainsthe rarityofthis case.Similarly, thiscase istheﬁrst casereportof bilateralosteoidosteomaofjaw.

Frequently,itaffectsyoungindividualsmainlythesecondand third decades of life. Jaw’s osteoid osteoma shows no gender predilectionincontrarytomalepredominanceofskeletalosteoid osteoma[7].Severepainistheprincipalsymptomasinthepresent case,whichatﬁrstisintermittentandbecomessevere,constant during the night. This nocturnal pain has the character to be

relieved by non-steroidal anti-inﬂammatory drugs (NSAIDs).

Generally, this symptom is attributed tochanges in thevessel pressurebeingregisteredbytheabundantinnervationpresentor todirectirritationofnerveﬁbersinornearthecalciﬁedfocus[2,8].

Radiographically,CTscanisthemostusefulpreoperativestudy indiagnosingandlocalizingofbonetumors.Thetypicalnidusof osteomaosteoidisaroundorovoidradiolucencylessthan1cmin diameter, having no or a small radiopacity in thecenter. Itis locatedattheintracortexorsubperiostealsite,andissurrounded by reactive sclerosis of the bone or sometimes with thick homogenous periosteal reaction. While, osteoblastoma is a radiolucent or mixed radiopaque lesion larger than 2cm in diameter,locatedinthemedullaryboneandcausingexpansionof theaffectedbone.Usually,surroundingreactivesclerosisisabsent, Fig.1.Imageofpatientshowingaswellingoftheleftangleofthemandible.

Fig.2.AxialCTrevealedcorticallocalizationofacalciﬁedmassontheleftangleof themandible,measuring2.48cm/2.01cm.

Fig.3.AxialCTrevealedaccidentallyasecondcalciﬁedmassontherightangleof themandible,measuring0.6cm.

Fig.4.Excisedspecimen.

T.Adoulyetal./InternationalJournalofPediatricOtorhinolaryngologyExtraxxx(2015)xxx–xxx 2

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exceptforathinborderofscleroticbone[9].Inthepresentcase,CT scan has detected one nidus located in the cortical bone;

approximately 10mm in diameter; in the left angle of the mandibleassociatedwithreactiveperiostealandreactivesclerosis.

Also,CThasdeterminedasecondbonemassontheotherangle withreactiveperiosteal.

Osteoid osteomas are classiﬁed into cortical, medullary or cancellous,andsubperiostealorperiosteal.Corticaltumorsarethe most common [10]. In the present case, osteoid osteoma is classiﬁedascorticaltype.

Intheliterature,thetreatmentofchoiceisacompleteexcision.

Thespontaneouspainimmediatelydisappearsaftersurgery.The localrecurrenceisduetothedevelopmentofasecondlesion,orto theresultofincompleteremovaloftheoriginallesion[11].The maincomplaintofourpatientwasmoderatetoseverepaininthe leftandtherightangleofmandible.Thedecisionwasacomplete excisionofbothlesions.Duringthefollow-upperiod,therewasno recurrence. Furthermore, there were no signiﬁcantradiological changesafter6months.

4. Conclusion

Osteoidosteoma of themandible is very rare. Furthermore, casesthatpresentedmultifocalosteoidosteomaarerarelybeen reported.Todatenomalignanttransformationhasbeenreported.

Inrecurrentlesionsitmustalwaysbekeptinmindthatthelesion could not be removed completely or there might be multiple osteoidosteomas.

Conﬂictsofinterest None.

Informedconsent

Informedconsentwasobtainedfromthefatherofourpatient.

References

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