Verruciform xanthoma of the palatal gingiva: a report of ... · Verruciform xanthoma of the palatal...
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292
Verruciform xanthoma of the palatal gingiva: a report of two cases
Da Jung Ryu1, Sang Hoon Lee1, Jong In Yuk2, Hyung Jun Kim3, Joing-Ki Huh1, Kwang-Ho Park1
1Department of Oral and Maxillofacial Surgery, Gangnam Severance Hospital, 2Department of Oral Pathology, Oral Cancer Research Institute, 3Department of Oral and Maxillofacial Surgery, College of Dentistry, Yonsei University, Seoul, Korea
Abstract (J Korean Assoc Oral Maxillofac Surg 2013;39:292-296)
Verruciform xanthoma (VX) is a rare, benign lesion that presents in the oral cavity, skin, or genital organs as a verrucous, papillomatous, or flat papule with varying colors. VX has indistinct clinical features, making histopathological examination necessary for a definitive diagnosis. Histologically, VX is characterized by parakeratosis, rete ridges with uniform depth, and an accumulation of the foam cells, which are also known as the “xanthoma cells”. These foam cells test positive for antibodies, such as CD-68 and vimentin; it is thought that VX foam cells are derived from the monocyte-macrophage lineage, and that VX’s pathogenic mechanism is partly related to an immune mechanism. Nevertheless, the pathogenesis of VX remains unclear. VX can be treated by surgical excision; other medical, chemical, and radiological treatments are not required postoperatively. Recurrence and malignant transformation of VX are rare. Two patients, each with a mass of unknown origin on the palatal gingiva, were presented at our clinic. Excisional biop-sies of the masses were performed for a histological diagnosis after clinical and radiological examinations. Histological examination confirmed a diag-nosis of VX in both cases.
Key words: Verruciform xanthoma, Mouth, Foam cells, Immune mechanism[paper submitted 2013. 8. 12 / revised 2013. 9. 24 / accepted 2013. 9. 26]
ThedevelopmentofVXisthoughttoberelatedtoanon-
specificreaction to localepithelial traumaoran immune
reaction.However,theetiologyandpathogenesisofVXare
notyetfullyunderstood6.VXcanbetreatedwithconserva-
tiveresection.Recurrenceandmalignanttransformationof
VXarerare,andthedisorderusuallyhasagoodprognosis3.
Sofar,onlyafewcasesofVXpresentingintheoralcavity
havebeenreported4.Wereport twocasesofVXthatpre-
sentedonthemaxillarypalatalgingivaofmalepatientsand
reviewtherelevantliteratureonthisdisorder.
II. Cases Report
1. Case 1
A37-year-oldmanvisitedourhospital(GangnamSever-
anceHospitalinSeoul,Korea)becauseofapoorlyhealing
woundonhispalate.Thepatientaccidentallydiscoveredthe
woundoneyearpriortohisfirstvisit.Althoughthepatient
didnotexperienceanypain,hedescribedthelesionasbeing
somewhatuncomfortableorsensitivewhenirritatedbyfood
orpalpation.Thepatientwasadvisedtoundergoabiopsy
eventhoughthelesiondidnotseemtoprogressandchange
I. Introduction
Verruciformxanthoma(VX)isarare,benignlesionthat
presentsintheoralcavity,skin,orgenitalorgans.Itgrows
slowly,usuallywithoutcausingpain,and itsappearance
resembleslocalepithelialhyperplasiasuchasverruciform
papules,papilla,andplaque1.ThecolorofVXcanvarybe-
tweenwhite,pink,andyellow,dependingonthethickness
oftheoverlyingepidermis.VXcontainsfoamcells,which
arelipid-containinghistiocytesfoundwithinthesubmucosal
substrates2,3.Thesefindingsaresimilartothoseobservedin
leukoplakia,papilloma,verrucavulgaris,condylomaacumi-
nata,andcertainmalignantlesions,anditisthusnecessaryto
differentiateVXfromtheselesionswithatissuebiopsy4,5.
CASE REPORThttp://dx.doi.org/10.5125/jkaoms.2013.39.6.292
pISSN 2234-7550·eISSN 2234-5930
Kwang-Ho ParkDepartment of Oral and Maxillofacial Surgery, Gangnam Severance Hospital, College of Dentistry, Yonsei University, 211, Eonju-ro, Gangnam-gu, Seoul 135-720, KoreaTEL: +82-2-2019-4560 FAX: +82-2-3463-4052E-mail: [email protected]
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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Copyright Ⓒ 2013 The Korean Association of Oral and Maxillofacial Surgeons. All rights reserved.
Verruciform xanthoma of the palatal gingiva
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theprobabilityofmalignancywasquitelow.Anexcisional
biopsy,ratherthananincisionalbiopsy,wasthenplanned
andperformedinordertoresectthetotallesionandobtaina
definitivediagnosis.Histopathologicalexaminationrevealed
apapillaryproliferationofthestratifiedsquamousepithelial
celllayer,whichwascoveredwithahyperkeratoticlayerof
uniformthickness,aboutthatofthestratumspinosum.(Fig.3.
A)Distinctivefoamcells,alsoreferredtoasxanthomacells,
aswellasmacrophageswithvacuolatedcytoplasmcouldbe
seenintheconnectivetissuelayerwhenmagnifiedundera
high-resolutionmicroscope.(Fig.3.B)
Afterthelesionwastotallyresected,thesurgicalsiteun-
derwentnormalsecondaryhealingprocesses.Thepatient
hadnocomplications,andtherewasnoevidenceofrecurrent
diseaseduringthethree-yearfollow-upperiod.(Fig.4)
2. Case 2
A35-year-oldmanwasreferredtoourclinicforevalua-
tionofalesionthatdevelopedonhishardpalate.Thepatient
becameawareofthelesiononemonthpriortohisfirstvisit.
Duringconsultation,hecomplainedthatthelesionbecame
sensitivewhenitcameincontactwithhotandsaltyfoods,but
heexperiencednootherspecificsymptoms.Thepatienthad
ahistoryofanunknownsexuallytransmitteddisease,which
hecontractedoneyearpriortohisfirstvisitandcompletely
recoveredfrombythetimeofconsultation.Inaddition,the
patienthada20-yearhistoryofheavysmoking.Noother
specificgeneraldiseasesorexceptionalmedicalhistorywere
noted.Duringtheclinicalexamination,whichwasconducted
duringhisfirstconsultation,asoftsessilemasswithaverru-
cous,papillarysurfaceresemblingastrawberrywasfoundon
thepalatalgingivaoftheleftmaxillarysecondpremolarand
firstmolar.Thelesionwas2.5×1.0cminsize,welldemar-
cated,andpinkishincolor.Itwasnotpainfulwhenpalpated.
Healsohadanormalresponseinthepercussion,mobility,
insize,shape,orposition.
Thepatientdidnotreportanysignificantpastmedicalhis-
tory,historyoftrauma,orexceptionalmedicalorfamilyhis-
tory.Hereporteda13-yearsmokinghistorybutnootherspe-
cificsocialhabits.Duringtheintraoralexamination,awell-
demarcatedsoftandpedunculatedpapulewithaverruciform
papillarysurfacewasfoundontheleftsideofthehardpalate.
Thelesionwasapproximately1.5cmindiameter,reddishin
color,andclinicallyasymptomatic.(Fig.1)Nootherintraoral
orskinlesionswereidentified.Duringtheradiologicalexam-
ination,completewithperiapicalandpanoramicradiographs,
nospecificfindingswereobserved.(Fig.2)Consideringthe
resultsof theclinicalandradiographicexaminations,we
identifiedmultipleprovisionaldiagnosesofthislesion,in-
cludingleukoplakia,papilloma,condylomaacuminatum,and
malignanciessuchassquamouscellcarcinomaorverrucous
carcinoma.Giventhelesion’ssize,history,well-demarcated
margin,andtheresultsofanecknodeexamination,however,
Fig. 2. Radiological examination of Case 1. A. Periapical view. B. A magni-fied view of orthopantogram. No specif-ing findings on these two radiograph images. Da Jung Ryu et al: Verruciform xanthoma of the palatal gingiva: a report of two cases. J Korean As-soc Oral Maxillofac Surg 2013
Fig. 1. Clinical aspect of the palate lesion in Case 1 on the first visit. A reddish papule with a rough verrucous surface and distinc-tive margin can be observed.Da Jung Ryu et al: Verruciform xanthoma of the palatal gingiva: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2013
J Korean Assoc Oral Maxillofac Surg 2013;39:292-296
294
Fig. 3. Histopathological examination of Case 1 after excisional biopsy. A. The tissue has a papillary appearance with hyperparakeratosis and uniform rete peg (H&E staining, ×40). B. High magnification (H&E staining, ×200) illustrates large foam cells (xanthoma cells, black arrows) in the connective tissue papillae.Da Jung Ryu et al: Verruciform xanthoma of the palatal gingiva: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2013
Fig. 4. Follow-up clinical photos of Case 1 representing a good secondary healing state. A. Two weeks postoperative. B. Three weeks postoperative. C. Five weeks postoperative. D. Four months postoperative. E. One year postoperative. Da Jung Ryu et al: Verruciform xanthoma of the palatal gingiva: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2013
Fig. 5. Histopathological examination of Case 2 at the diagnostic incisional biopsy. A. Papillary surface with parakeratosis (H&E staining, ×100). B. Xanthoma cells (black arrows) (H&E staining, ×400).Da Jung Ryu et al: Verruciform xanthoma of the palatal gingiva: a report of two cases. J Korean Assoc Oral Maxillofac Surg 2013
Verruciform xanthoma of the palatal gingiva
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(between40and60yearsofage),butitcanalsodevelopin
otheragegroups.Further, ithasnospecificsexpredomi-
nance4,5.Theoralcavityandthegenitalmucosaarethemost
affectedregions,andVXusuallyappearsasasinglelesion.
VXcanariseinanypartoftheoralcavity,althoughthemost
commonlyaffectedsitesare,thegingiva,hardpalate,tongue,
andbuccalmucosa, inorderofcommonality4.Although
multipleVXlesionshavebeenreportedinseveralpatient
groupswithsystemic lipidmetabolicabnormalities, there
has,ingeneral,beennodefinitecorrelationbetweenVXand
systemicdiseases8.Furthermore,VXdoesnotseemtohavea
specificrelationshipwithenvironmentalfactorssuchasahis-
toryofsmokingordrinkingortheuseofprostheticssuchas
dentures5.
Thenameofthediseaseimpliesitsclinicalcharacteristics
aswellasitshistologicaldiagnosis.VXthatdevelopswithin
theoralmucosapresentsclinicallyasasingleindolentlesion
withasurfaceofflatpapillaryorverruciformprotrusions.
Suchlesionsareusuallyabouttwocmindiameterandcan
havevariouscolorsincludinggray,pink,andyellowdepend-
ingonthethicknessandpropertiesoftheoverlyingepider-
mis1-5.Itisessentialtoobtainatissuebiopsyforadefinitive
diagnosisofVXbecauseitisdifficulttoclinicallydifferenti-
atebetweenVXandotherdiseases involvingverruciform
surfaces.Otherconditionswithsimilarclinicalfeaturesthat
shouldbeconsideredareleukoplakia,papilloma,verrucavul-
garis,condylomaacuminata,andmalignantlesionssuchas
squamouscellcarcinomaorverrucouscarcinomawithver-
ruciformsurfaces9,10.
Pathohistologically,VXpresentswithrelativelyspecific
characteristics.Ingeneral,hematoxylin-eosinstainingshows
aparakeratotic layer thatextendsto thepapillarysurface,
whiletheepithelialretepeghasagenerallyeventhickness
withaprominentextension into theunderlyingconnec-
tivetissue.Amarkedincreaseinepithelialcelldivisionor
pseudo-epithelialhyperplasiaisnotobserved.Infiltrationof
chronicinflammatorycellsthatcontainfewneutrophilsand
cellgroupsconsistingofseveralsmallandlargecellswith
vacuolatedcytoplasmaredominant inthedermalconnec-
tivetissuelayerbetweentheextendedretepegs.Theseare
specificfoamcellscalledxanthomacells,whichcouldbethe
pathologicalandhistologicalcharacteristicsofadefinitive
diagnosisforVX2.Whenstainedbyscharlachred,xanthoma
cellspresentwithanorange-redcolortypicaloflipids,and
severalsmalland large lipidgranulescanbeseen in the
cytoplasmwhenhistologyslidesareviewedunderahigh-
resolutionmicroscope.OtherfeaturesofVXsuchasswell-
andvitalitytestsofhisteeth,however;thepalatalaspectof
his leftmaxillaryfirstmolarshowedmildgingivalreces-
sion.Theradiologicalexamination,includingperiapicaland
panoramicradiographs,didnotyieldspecificresults.Further,
therewasnoenlargementorindurationofhisbilateralneck
lymphnodesonpalpation.Thoughwecouldnotobtainmore
informationabouthismedicalhistoryinregardtohissexual-
lytransmitteddisease,wesuspectedarelationship.Wediag-
nosedthelesionwithdifferentialsthatincludedsyphilisand
malignanciessuchassquamouscellcarcinomaorverrucous
carcinoma.Anincisionalbiopsyforadefinitivediagnosis
wasperformedoneweekaftertheinitialexamination.During
histopathologicexamination,histologyslidesobtainedfrom
thebiopsyshowedapapillarysquamousepithelialcelllayer
coveredwithparakeratoticsurface(Fig.5.A)andxanthoma
cellsinthesubepithelialconnectivetissuelayer(Fig.5.B),
distinctivefeaturesofVX.Conservativesurgicalresection
wasperformedaftertheresultsofthebiopsywereconfirmed.
Postoperativefollow-upexaminationshowedawell-healed
surgicalsite,andthepatienthadnocomplaints.
Fouryearsafterthisresection,thepatientreturnedtoour
clinicfortherecurrentappearanceofthelesionatthesame
site.Thelesionappearedtohavegrowntoasimilarsize,and
itsshapewasalsosimilartotheoneresectedpreviously.Fur-
ther,therecurringmassaffectedthesamepartofthebuccal
gingivaaroundhisleftmaxillarysecondpremolarandfirst
molar.Onceaclinicaldiagnosisofarecurrentlesionofthe
pre-existingVXwasmade,awidesurgicalresection,which
includedseeminglynormaltissuessurroundingthelesion,
wasperformed.Theresultsofthehistopathologicexamina-
tionoftherecurrentlesionwerethesameasthatoftheinitial
lesion.Thepatienthasbeenfollowed-upatregularintervals
offewerthansixmonthsforthepasttenyears,andtherehas
beennoevidenceofVXrecurrence.
III. Discussion
VXisarelativelyrarebenignlesionthatwasfirststudied
anddefinedbyShafer1in1971.Sincethen,thepresenceof
VXintheoralcavityhasbeenreportedinabout300cases4,
whileonlyfivecasesofVX-relatedskinlesionshavebeen
reportedinSouthKorea6,7.Althoughtherehavebeennodefi-
nitestudiesontheincidenceofthisdisease,aprevalenceof
approximately0.025%to0.094%hasbeenreported4,5.To
date,onlyabouttencasesofVXhavebeenreportedinAsian
andAfrican-Americanstudies,withthemajorityofpatients
beingCaucasian1-5.VXcommonlyoccursduringmiddleage
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296
nologicallycompromisedstatus,systemicdiseases,andVX,
wedeemitnecessarytoimprovetheknowledgeofVXand
havethereforereportedtwocasesofVXtreatedatourclinic.
References
1. ShaferWG.Verruciformxanthoma.OralSurgOralMedOralPathol1971;31:784-9.
2. ZegarelliDJ,Zegarelli-SchmidtEC,ZegarelliEV.Verruciformxanthoma.Furtherlightandelectronmicroscopicstudies,withtheadditionofathirdcase.OralSurgOralMedOralPathol1975;40:246-56.
3. vanderWaalI,KerstensHC,HensCJ.Verruciformxanthomaoftheoralmucosa.JOralMaxillofacSurg1985;43:623-6.
4. PhilipsenHP,ReichartPA,TakataT,OgawaI.Verruciformxan-thoma--biologicalprofileof282orallesionsbasedonaliteraturesurveywithninenewcasesfromJapan.OralOncol2003;39:325-36.
5. YuCH,TsaiTC,WangJT,LiuBY,WangYP,SunA,etal.Oralverruciformxanthoma:aclinicopathologicstudyof15cases.JFormosMedAssoc2007;106:141-7.
6. NamYH,OhCW.Acaseofverruciformxanthomapresentingasleukoplakiaonthelowerlip.KoreanJDermatol2006;44:508-11.
7. ChoiYH,MoonYJ,LeeYW,WonJY,SongES.Acaseofmul-tipleverruciformxanthomaoforalcavityandgastrointestinaltract.KoreanJDermatol2002;40:162-5.
8. TravisWD,DavisGE,TsokosM,LebovicsR,MerrickHF,MillerSP,etal.Multifocalverruciformxanthomaoftheupperaerodiges-tivetractinachildwithasystemiclipidstoragedisease.AmJSurgPathol1989;13:309-16.
9. MeteO,KurkluE,BilgicB,BekaH,UnurM.Flat-typeverruci-formxanthomaofthetongueanditsdifferentialdiagnosis.Derma-tolOnlineJ2009;15:5.
10. NevilleBW,DammDD,AllenCM,BouquotJE.Oral&maxillo-facialpathology.2nded.Philadelphia:WBSaunders;2002.
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12. ShahrabiFarahaniS,TreisterNS,KhanZ,WooSB.Oralverruci-formxanthomaassociatedwithchronicgraft-versus-hostdisease:areportoffivecasesandareviewoftheliterature.HeadNeckPathol2011;5:193-8.
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ingofthemitochondria,extensionoftheGolgiapparatus,
expansionoftheendoplasmicreticulum,anddestructionof
thejunctionalcomplexescanalsobeobservedunderahigh-
resolutionmicroscope.HistologicalfeaturesofVX,includ-
inghyperkeratosis,infiltrationofneutrophils,andfoamcells,
areregularlyseeninotherinjuredcells,andit istherefore
thoughtthatVXstartswithepitheliumatrophyanddestruc-
tionandthenproceedstothegenerationoffoamcells11.This
premiseissupportedbyreportsthatshowthatVXisassoci-
atedwithdiseasessuchaslichenplanus,lupuserythemato-
sus,vesicularepidermolysisbullosa,pemphigusvulgaris,and
graft-versus-hostdisease8,11,12.
In2005,Huetal.13conductedimmunohistochemicaltests
usingvariousmonoclonalantibodiesinanefforttoidentify
thepathogenesisofVX.Theauthorsconcludedthatfoam
cellsinVXtestedpositiveforspecificantibodiesofCD68
andvimentin,andthatmacrophagesandimmunecellsinthe
monocytesystemfulfilledaroleintheimmunemechanism.
However,Huetal.13couldnotidentifyaclearpathogenesis
forVX.Thusfar,variousstudieshavepresentedpossible
factorsthatcouldcauseVX,includingmicrobialcommunity,
humanpapillomavirus,lymphedema,genetictendency,and
dermaltraumawithaninflammatoryresponse11,13,14.Never-
theless,toourknowledge,nopublishedstudyhasidentified
significantcorrelationsbetweenthesefactorsandtheoccur-
renceofVX.Thus,itisnecessarytoinvestigatesuchcorrela-
tionsinthefuture.
VXcanbetreatedwithconservativeresection,anditdoes
notrequiremedical,chemical,orradiologicaltreatmentafter
surgery.RecurrentVXandmalignant transformationsare
rare.Inthiscasepresentation,wereportonecaseofrecur-
rencefouryearsafterconservativesurgicalresection.After
thesecondsurgicalprocedure,VXdidnotrecurinafollow-
upperiodlongerthantenyears.Thereportedcasesalsoshow
thattheprognosisofVXisrelativelygood1-5,9,10,15.
AsVXisararebenigntumor,generalpractitionersand
evenspecialistssuchasoralandmaxillofacialsurgeonsare
rarelyawareofthisdisease.Thusfar,therehavebeenvery
fewreportsofVX.Consideringthattherearealimitednum-
berofstudiesinvestigatingtherelationshipamonganimmu-