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Unusual Odontogenic

Keratocyst of the Maxillary Sinus


ConstantinosHoupis,DDS,PhD,MSc,
KonstantinosI.Tosios,DDS,PhD,StavroulaMerkourea,DDS,
StylianosKrithinakis,DDS,NicolaosNikitakis,DDS,PhD,
AlexandraSklavounou,DDS,PhD,MSc
Abstract:
Anodontogenickeratocystthaterodedintothesinus
throughthemaxillaryboneandoccupiedit,showedreplacementof
thesinusrespiratoryepitheliumbylesionalepithelium,andwas
associatedwithfungalrhinosinusitisispresented.Areviewofthe
literaturedisclosedthatepithelialreplacementhasbeendescribedin
2previouscasereports,althoughthereisnoreportonthecoexis
tenceofodontogenickeratocystwithfungalrhinosinusitis.
KeyWords:
Odontogenickeratocyst,maxilla,fungalrhinosinusitis

dontogenickeratocyst(OKC)isestablishedasadistincten
titybecauseofitsspecificmicroscopicfeaturesandaggres
sivebehaviorthatmanifestswithinfiltrationofadjacentanatomic
FromtheDepartmentofOralPathologyandSurgery,DentalSchool,
NationalandKapodistrianUniversityofAthens,Athens,Greece.Drs.
Houpis,Merkourea,andKrithinakisareinprivatepractice.
ReceivedJune15,2010.
AcceptedforpublicationJuly4,2010.
AddresscorrespondenceandreprintrequeststoConstantinosHoupis,
DDS,PhD,MSc,PlEsperidon2A,16674Glyfada,Greece;
Email:kchoupis@otenet.gr
PresentedasaposterattheAmericanAcademyofOral&Maxillofacial
Pathologyannualmeeting,May16
Y
20,2009,Montreal,Canada.
Theauthorsreportnoconflictsofinterest.
Copyright
*
2011byMutazB.Habal,MD
ISSN:10492275
DOI:10.1097/SCS.0b013e31820747b7

The Journal of Craniofacial Surgery

&
Volume 22, Number 2, March 2011
Brief Clinical Studies
*
2011MutazB.Habal,MD

721

Copyright 2011 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
structuresandhighrecurrencerate.Thisbehaviorisemphasized
bytheintroductionoftheterm
keratocysticodontogenictumor

intherecentclassificationoftheWorldHealthOrganization,
1

althoughitsneoplasticnatureisdebated.WedescribeanOKCthat
erodedintothesinusthroughthemaxillaryboneandreplacedthe
sinusrespiratoryepithelium,whileitwasassociatedwithfungal
rhinosinusitis.

CLINICAL REPORT
A38yearoldmanpresentedwithintermittentpain,swelling,and
afeelingoffullnessandpressureontheleftsideofhisfacethat
hehadfirstnoticedabout1yearagoandgotworseduringscuba
diving.Hismedicalhistorywassignificantforchronicrhinosinus
itisthathadrepeatedlybeentreatedwithadministrationofanti
bioticsandlocalcorticosteroids.Hewasotherwisehealthyandnot
inanyotherkindofmedication.
Clinicalexaminationrevealedahard,nonfluctuant,andslightly
tenderswellingcoveredbynormalmucosainthemaxillaryvesti
bule,distanttothefirstmolartooth.Regionallymphnodeswerenot
palpable.Accordingtothereferringdentist,theassociatedteeth
werevital.Panoramicradiographshowedawellcircumscribedra
diolucentlesionapicaltothefirstmolartooth(Fig.1).
AprovisionaldiagnosisofOKCwasmade,andanincisional
biopsywasperformedunderlocalanesthesia.Intraoperatively,the
cysticcavitywasfoundtocontainawhitishmaterial.Microscopic
examinationof5
K
mthick,formalinfixed,andparaffinembedded
tissuesectionsshowedconnectivetissuelinedbyauniformlythin
parakeratinizedepitheliumwithoutreteridgesthathadacorru
gatedparakeratinizedsurfaceandabasallayercomposedofco
lumnarcellswithreversenuclearpolarity(Fig.2).Focalseparation
oftheepitheliumfromtheconnectivetissuewasseen.Thecon
nectivetissuewasmildlyinfiltratedbyinflammatorycells,pre
dominantlylymphoplasmacytes.ThediagnosiswasOKC.
Acomputedtomographyscanrevealedadiffuseradiopacity
mainlytowardtheflooroftheleftmaxillarysinus.Boneresorption
wasevidentattheanterolateralwallofthemaxillaclosetothe
alveolarprocess(Fig.3).Radiopacityofthesinuswithmicrocal
cificationsormetallicdensespotswereinterpretedasconsistent
withaspergillomas.
Thecystwasenucleated,thesinuscleanedthroughaCaldwell
Lucapproach,andadrainwasplacedthrougharhinoantrostomy.
Thepatientwasadministeredamoxicillin500mgplusclavulanic
acid125mg,3timesdaily,for5days.Thedrainwasremovedafter
72hours;hispostoperativerecoverywasuneventful,and19months
afteroperation,heisfreeofdisease.
Macroscopically,thecystmeasuredabout3.5
2
1cm.
MicroscopicexaminationconfirmedthediagnosisofOKC.
Focally,theepitheliumshowedincreasedcellularityanddrop
shapedreteridges,whereasthecellsinthebasalthirdhadlarge
andhyperchromaticnuclei(Fig.4).Mitoseswerenotidentified.
Inaddition,therewasafocusofabrupttransitionofthecystic

epitheliumtorespiratorypseudos
tratifiedcolumnarepithelium
thatgavetheimpressionofactivereplacementorpushing
(Fig.5).Theconnectivetissueofthecysticwallwasvascular,
edematous,withfociofcalcificati
ons,cholesterolcrystals,and
mildlymphoplasmacyticinfiltration,whereasnogranulomatous
FIGURE 1.
Panoramic radiograph shows well-circumscribed
radiolucent lesion apical to the first molar tooth.
FIGURE 2.
Connective tissue lined by a uniformly thin
parakeratinized epithelium without rete ridges. Notice the
corrugated parakeratinized surface and a basal layer
composed of columnar cells with reverse nuclear polarity
(hematoxylin-eosin stain, original magnification
400).
FIGURE 3.
A, Computed tomography scan, coronal view,
reveals diffuse radiopacity mainly toward the floor of the left
maxillary sinus, bone resorption at the anterolateral wall of the
maxilla close to the alveolar process, and microcalcifications
(arrows). B, Computed tomography scan, sagittal view,
reveals diffuse radiopacity mainly toward the floor of the left
maxillary sinus and microcalcifications (arrows).
FIGURE 4.
Odontogenic keratocyst epithelium shows
increased cellularity, drop-shaped rete ridges, and cells with
hyperchromatic nuclei in the basal compartment
(hematoxylin-eosin stain, original magnification
400).
BriefClinicalStudies
The Journal of Craniofacial Surgery

&
Volume 22, Number 2, March 2011

722

*
2011MutazB.Habal,MD
Copyright 2011 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
reaction,fibrosis,ornecroticfociwereevident.Periodicacid
Schiffstainrevealedfungalhyphaeandsporesinthemucosal
connectivetissue,closetobutnotinfiltratingvessels(Fig.6).
FinaldiagnosiswasOKCwithyeast.
Acompletebloodcountwasnotsuggestiveofhematologic
diseaseordiabetesmellitus,andanHIVtestwasnegative.

DISCUSSION
SinusinvolvementbyOKCsisestimatedtooccurinlessthan1%of

thecases,
2

butreplacementofrespiratoryepitheliumwithOKC
epitheliumisunusual.Yamazakietal
3

describedacaseofanOKC
ina39yearoldwomanthatinvolvedanimpactedmaxillarypre
molar,expandedtothemaxillarysinus,andfinallyreachedthe
inferiornasalmeatus.ReplacementoftheOKCliningbyrespiratory
epitheliumwasattributedtotheproximityofthecysttothenasal
cavity.AbruptchangeofOKCliningtorespiratoryepithelium,as
seeninourpatient,wasreportedbyVencioetal
4

ina27yearold
woman.Thecystwasassociatedwithanimpactedrightsecond
maxillarymolarandalsoinvadedthesinusfloor.
Thismicroscopicfindingisreminiscentoftheextensionof
intraepithelialneoplasiaoftheoralcavity
5,6

andothercarcinomas
7

alongadjacentductalepitheliumbasementmembrane.Ductalor
glandularinvolvementisconsideredanimportantpathwayof
spreadofcarcinomas.Inourcase,weassumethattheOKCepi
theliumcreptonthesinuswallreplacingthenormalrespiratory
epitheliumandextendedtoinvolvemostoftheantralcavity.Thus,
weagreewithVencioetal
4

inthatreplacementisconsistentwith
theinfiltrativebehavioroftheOKCreflectedinitsclassification
asaneoplasm.Dysplasticfeatures,asseeninthepresentcase,
areoccasionallydescribedinOKCs,buthavenotbeenassociated
withanaggressivegrowth.
Ourpatienthadahistoryofchronicrhinosinusitisthatisthe
mostcommonformofrhinosinusitis,estimatedtoaffectabout
20%ofthepopulation.
8

Radiologicalevidenceofsinusopaci
ficationwithcalcificationswasconsistentwithfungusballsor
aspergillomas,definedasnoninvasiveaccumulationsofdensecon
glomerationoffungalhyphaein1sinuscavity.Histologicalevi
dence,however,ofsporesandhyphaeinthemucosalconnective
tissueisdiagnosticofaninvasivefungalrhinosinusitis,inparticular
chronicinvasivefungalrhinosinusitis.Thisformisusuallyassoci
atedwith
Aspergillusfumigatus
andmayappearinpatientsunder
corticosteroidtreatmentwhoaresubtlyimmunocompromised.
NoothercaseofOKCassociatedwithfungalrhinosinusitis
andfungusballswasfoundintheliterature.Inourpatient,theroot
canaltherapiesinthecentralincisorsandrightlateralincisorwere
notclosetothesinus;thus,arolefortheheavymetalsfromroot
fillingmaterials,inparticular,zincfromzincoxide
Y
containing
materials,donotseemprobable.Theblockageoftheostiumbythe

OKC,however,mayhaveresultedintheobstructionofsinusven
tilationandthecreationofanaerobicconditionsthatallowedger
minationoffungi,whichprobablybecameinvasivebecauseofthe
localimmunosuppressioncausedbytheprolongedcorticosteroid
treatment.
9,10

Asourpatientwasprovedtobeimmunocompetent,thecom
pletesurgicalremovalofthecystandthesinusdrainageandaera
tionachievedwiththerhinoantrostomywereconsideredcurative,
bothfortheOKCandthefungalrhinosinusitis.
11

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