Case report: HIV negative isolated scrotal Kaposi's sarcoma

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International

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of

Surgery

Case

Reports

jo u r n al ho me p a g e :w w w . c a s e r e p o r t s . c o m

Case

report:

HIV

negative

isolated

scrotal

Kaposi’s

sarcoma

Hamid

Ozmen

,

Dursun

Baba,

Coskun

Kacagan,

Ali

Kayikci,

Kamil

Cam

DepartmentofUrology,DuzceUniversitySchoolofMedicine,Duzce,Turkey

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r

t

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Articlehistory: Received5July2014

Receivedinrevisedform11October2014 Accepted6November2014

Availableonline13November2014 Keywords:

Casereport Kaposi’ssarcoma Scrotum

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INTRODUCTION:Kaposi’ssarcoma(KS)isarareangioproliferativedisorderofthevascularendothelium. ThedevelopmentofKSrequiresHumanHerpesVirus8(HHV-8)infection.AnassociatedHIVinfection isusuallyseen.IsolatedscrotalKShasrarelybeenreported.Inthisarticle,wepresentacaseofKSthat primarilyinvolvedthescrotuminaHIVnegativepatient.

PRESENTATIONOFCASE:A71-yearoldmalepatientadmittedtotheoutpatientdepartmentduetonodular lesionsonthescrotum.Thepatientdeclaredthattheselesionswerepresentfornearly5years.Past medi-calhistoryrevealedthatheunderwentleftthoracotomyandupperlobectomyin2006foradenosquamous lungcarcinoma.Then,hereceivedasinglecycleofadjuvantchemotherapyconsistedofdocetaxeland cisplatin.Physicalexaminationrevealed3blacksmallnodulesonthescrotum.Theanti-HIVtestwas negative.Allscrotallesionsweresurgicallyexcised.ThepathologicalinvestigationrevealedKSofthe lymphangioma-liketype.

DISCUSSION:ThepathogenesisofKShasstillnotbeenclearlyelucidated.However,itisknownthat allformsofKSareassociatedwithHHV-8infections.Adefectinimmunesystemwasalmostalways necessary.Therefore,KSisusuallyassociatedwithHIVinfection.KSofthepenishasbeenreportedinHIV negativepatients.VeryfewcasesofscrotalKShavebeenpresented.Inarecentreview,only1patienthad scrotalKSoutof32caseswithHIVnegativeKS.Inourcase,thepatientreceivedacycleofchemotherapy thatmightaffecthisimmunesystem.Thelymphangioma-liketypeisacommonmorphologicalsub-type. Whilelymphedemasarecommonlyobservedinthissub-type,noedemainthelymphswaspresentin ourcase.

CONCLUSION:ClassicalKSisgenerallyobservedinthelowerextremities,itcanrarelyaffectscrotalskinas isolatedlesions.Therefore,acarefulphysicalexaminationshouldalsoincludescrotumforthesepatients.

©2014TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/3.0/).

1. Introduction

Kaposi’ssarcoma(KS)is arareangioproliferative disorderof thevascular endothelium. Thedevelopment of KSrequires the presenceofaHumanHerpesVirus8(HHV-8)infection.Astrong associationwithHIVinfectioniswellknown.Theclassicalformwas firstdefinedas“idiopathicmultiplepigmentedsarcoma”byMoritz Kaposiin1872.1 _{Theskinlesionsareclassicallycharacterizedby}

macules,plaquesandnodulesthatareofapurple,red,blue,dark brownorblackappearance.WhileKSprimarilyaffects mucocuta-neoustissues,itcanalsoaffectinternalorgans.ClassicalKSmostly affectsthelowerextremities.PenileKSisrelativelycommon,while

∗ Correspondingauthorat:DepartmentofUrology,DuzceUniversitySchoolof Medicine,Konuralp81620,Duzce,Turkey.Tel.:+903805421390;

fax:+903805421387.

E-mailaddress:[email protected](H.Ozmen).

isolatedscrotalKShasrarelybeenseen.2_{Inthisarticle,wepresent}

acaseofscrotalKSinaHIVnegativepatient.

2. Casereport

A71-yearoldmalepatientadmittedtotheoutpatient depart-mentdue toblack nodularlesionsonthescrotum. Thepatient declaredthattheselesionswerepresentfornearly5years.Past medical history revealed that he underwent left thoracotomy and upper lobectomy in 2006 for adenosquamous lung carci-noma.Then,hereceivedasinglecycleofadjuvantchemotherapy consistedofdocetaxelandcisplatin.Thechemotherapywas dis-continued due to theside effects. No recurrence was detected regardingthelungcancer.Thescrotallesionsappeared2months followingthechemotherapy.Physicalexaminationrevealed3black nodules onthe scrotum, each one beingnearly 0.5cm×0.5cm in dimension(Fig.1). No othersimilar lesionswere seen else-where.Routinelaboratorytestswereallnormal.Theanti-HIVtest (chemiluminescence)wasnegative.Hehadabdominalcomputed

http://dx.doi.org/10.1016/j.ijscr.2014.11.019

2210-2612/©2014TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense (http://creativecommons.org/licenses/by-nc-nd/3.0/).

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H.Ozmenetal./InternationalJournalofSurgeryCaseReports5(2014)1086–1087 1087

Fig.1.Threeblacknodulesonthescrotum.

tomographyforthefollow-upoflungcancershowingnospecific abnormalityunlessanincreaseinthethicknessofthestomachwall. Agastroscopywasthenperformedrevealingnolesions.All scro-tallesionsweresurgicallyexcised.Thepathologicalinvestigation revealedKSofthelymphangioma-liketype.

3. Discussion

In classical KS, lesions are primarily located on the lower extremities, and the involvement of external genitalia is uncommon.3_{AlthoughcasesofHIVnegativepatientsinwhichthe}

penisisprimarilyinvolvedhavebeenreported,veryfewcasesof scrotalKShavebeenpresented.Inarecentstudy,only1patienthad scrotalKSoutof32caseswithnon-HIVKS.2_{Also,itwasunclearthat}

thissinglecaseofscrotalKSwasassociatedwithotherlocationsor not.2_{PenileKShasbeenreportedmoreextensively.Vyasetal.first}

reportedanisolatedcaseofscrotalKSin1976.4 _Then,onlytwo

casesofisolatedscrotalKSwerepresented.5,6_{Ourcaserepresents}

the4thcaseuptoourknowledge.

Thepathogenesis ofKShasstill not beenclearlyelucidated. However,itisknownthatallformsofKSareassociatedwithHHV-8 infections.Adefectinimmunesystemwasalmostalways neces-sary.Therefore,KSisusuallyassociatedwithHIVinfection.Inour case,thepatientreceivedacycleofchemotherapythatmightaffect hisimmunesystem.

Thelymphangioma-liketypeisacommonmorphological sub-type.Whilelymphedemasarecommonlyobservedinthis sub-type,noedemainthelymphswaspresentinourcase.

AGreekstudyreportedasymptomaticstomachinvolvementin 82%ofpatientswithclassicalKS.7_{Theysuggestroutineendoscopy}

fornewlydiagnosedKSpatients.Ourcasehadnosystemiclesions onendoscopyandradiologicalimaging.Detailedphysical exami-nationsalsorevealednootherskinlesions.

StudiesconductedforthetreatmentforKSaregenerally per-formed on HIV patients. Unfortunately, there is currently no treatmentmethodcapableoferadicatingHHV-8.Forthisreason, themaingoalofKStreatmentistoalleviatethesymptomsofthe disease,toreducethesizeandnumberofcutaneousandvisceral

lesions,andtoreduceanddelaytheprogressionofthedisease.8

Despitetheuseofvariouslocalandsystemictreatmentmethods forclassicalKS,thereisnostandardtreatmentprocedure admin-istered tocaseswithgenitalKS.Atotal of19caseswithpenile KSwerereported.9 _{Localexcisionwasperformedon9patients,}

circumcision was performed on one patient, and radiotherapy, chemotherapyorIFN-alphatreatmentinadditiontolocalexcision wasperformedon3patients.Duringthefollow-upperiod,only4 caseswithlocalexcisionrelapsed.Vyasetal.performedtotal scro-tectomyandbilateralorchiectomyintheirreportwithscrotalKS.4

Wepreferredlocalexcisionforourpatient,sincetheselesionswere smallandstableforabout5years.Norecurrencewasobservedafter 2yearsoffollow-up.

Inconclusion, classicalKSisgenerallyobservedin thelower extremities, it canrarely affect scrotal skin as isolated lesions. Therefore,acarefulphysicalexaminationshouldalsoinclude scro-tumforthesepatients.

Conflictofinterest

Thereisnoconflictofinterest.

Funding

None.

Ethicalapproval

Awritteninformedconsentwasobtainedfromthepatient.

Authorcontributions

Hamid Ozmen: data collections, writing; Dursub Baba:data collections;CoskunKacagan:datacollections;AliKayikci:study design,datacollections;KamilCAM:studydesign,writing.

References

[1].BraunM.Classicsinoncology.Idiopathicmultiplepigmentedsarcomaoftheskin byKaposi.CACancerJClin1982;32:340–7.

[2].RescignoP,DiTrolioR,BuonerbaC,DeFataG,FedericoP,BossoD,etal. Non-AIDS-relatedKaposi’ssarcoma:asingle-institutionexperience.WorldJClinOncol 2013;10:52–7.

[3].AktasE,GülerE,Utas¸S,DenizK,OrhanO,YıldızOG.PenileKaposi’ssarcomain anHIVnegativemalepatient.Turkderm2008;4(2):131–3.

[4].VyasS,ManabeT,HermanJR,NewmanHR.Kaposi’ssarcomaofscrotum.Urology 1976;8:55–82.

[5].JohnsonDE,ChicaJ,RodriquezLH,LunaM.Kaposi’ssarcomapresentingasscrotal ulcerations.Urology1977;9(June(6)):686–8.

[6].SerranoC,SánchezG,delMarSerranoM,LinaresJ,DulantoC,NaranjoR.Nódulos yplacasvioláceasenescrotoymuslo.ActasDermosifiliogr2005;96(2):127–9.

[7].BalachandraB,TunitskyE,DawoodS,HingsI,MarcusVA.ClassicKaposi’ssarcoma presentingfirstwithgastrointestinaltractinvolvementinaHIV-negativeInuit male–acasereportandreviewoftheliterature.PatholResPract2006;202:623–6.

[8].KoliosG,KaloterakisA,FiliotouA,NakosA,HadziyannisS.Gastroscopic find-ingsinMediterraneanKaposi’ssarcoma(non-AIDS).GastrointestEndosc1995;42: 336–9.

[9].FatahzadehM.Kaposisarcoma:reviewandmedicalmanagement.OralSurgOral MedOralPatholOralRadiol2012;113:2–16.

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