CASE
REPORT
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OPEN
ACCESS
InternationalJournalofSurgeryCaseReports5(2014)1086–1087ContentslistsavailableatScienceDirect
International
Journal
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
i
c
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f
o
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:hamidozmen@hotmail.com(H.Ozmen).
isolatedscrotalKShasrarelybeenseen.2Inthisarticle,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/).
CASE
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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.3AlthoughcasesofHIVnegativepatientsinwhichthe
penisisprimarilyinvolvedhavebeenreported,veryfewcasesof scrotalKShavebeenpresented.Inarecentstudy,only1patienthad scrotalKSoutof32caseswithnon-HIVKS.2Also,itwasunclearthat
thissinglecaseofscrotalKSwasassociatedwithotherlocationsor not.2PenileKShasbeenreportedmoreextensively.Vyasetal.first
reportedanisolatedcaseofscrotalKSin1976.4 Then,onlytwo
casesofisolatedscrotalKSwerepresented.5,6Ourcaserepresents
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.7Theysuggestroutineendoscopy
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
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[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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