Sentinel lymph node biopsy in porocarcinoma: A case reports

InternationalJournalofSurgeryCaseReports53(2018)196–199

ContentslistsavailableatScienceDirect

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

Series

Sentinel

lymph

node

biopsy

in

porocarcinoma:

A

case

reports

Simona

Reina

_,

_Denise

_Palombo

_,

_Alexandru

_Boscaneanu

_,

_Nicola

_Solari

_,

Sergio

Bertoglio

_,

_Luca

_Valle

_,

_Ferdinando

_Cafiero

a_{DepartmentofSurgery,ChirurgiaI-OspedalePoliclinicoSanMartinoGenoa,Italy}

b_{DepartmentofSurgicalSciencesandIntegratedDiagnostics(DISC)GenoaUniversity,Italy}

c_{DepartmentofAnatomicPathology,OspedalePoliclinicoSanMartinoGenoa,Genoa,Italy}

a

r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received28August2018

Accepted13October2018

Availableonline31October2018

Keywords: Porocarcinoma

Malignantskinlesions

Sentinelnodebiopsy

Eccrinecarcinomas

a

b

s

t

r

a

c

t

INTRODUCTION:Eccrineporocarcinoma(EPC)isaslow-growingcarcinomaarisingfromtheeccrinesweat glands.Basedonitsclinicalpresentationitcanbeconfusedwithmalignantandbenignskinlesions, both.Histologicalexaminationisessentialtoformulateacorrectdiagnosis.Surgicalexcisionwithclear marginsisthestandardtherapeuticapproachwhiletheroleofsentinellymphnodebiopsy(SNLB)remains controversial.

CASEPRESENTATION:TheAuthorsreporttwocasesofEPCofthelowerlimbsoccurredintwowomen. PatientsweretreatedbywidesurgicalexcisionofthelesionandSNLB.6monthsfollow-upwasdisease freeforbothpatients.

CONCLUSION:Althoughararecutaneoustumor,EPChastobetakenintoaccountinthedifferential diagnosisofmalignantskinlesionsbecauseofitspossibleloco-regionalaggressivenessandrelated mor-bidity.Amongtheavailabletreatmentoptions,surgicalexcisionisconsideredthestandardapproach whereastheroleofSNLBiscontroversialalthoughtheAuthorsdiscussapossibleusefulnessforstaging anddiagnosis.

©2018TheAuthors.PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

1. Introduction

Eccrine porocarcinoma (EPC), first described by Pinkus and Mehreganin1963[1],isarareformofskincancer.Its presenta-tionsveryoftenmimicsacutaneouslesionsimilartootherforms ofbenignandmalignantcutaneousneoplasms[1,2].Accurate diag-nosis,optimaltreatmentandprognosisofEPCarestillchallenging duetoscantliteraturereports.Eccrinecarcinomasmayhavean elevatepresenceof regionallymphnodemetastasis,thussome authorshaveadvocatedSLNBforallorsomepatients,butitsutility forstagingpurposesremainsunknown.

WereporttwocasesofEPCinwhichthesentinellymphnode biopsy(SLNB)wasperformed[17].

∗ Correspondingauthor.

E-mailaddress:sergio.bertoglio@unige.it(S.Bertoglio).

2. Casepresentation

2.1. Case1

DuringAugust2017,a64yearswomanwasseenatour depart-mentafterapreviouscutaneouslesionexcisionwithanhistological diagnosis of porocarcinoma of the left thigh. The histological examinationrevealedaporoidneoplasmextendingintothedeep dermis to the level of the dermal-subcutaneous junction with a thickness of5.4mm,10–12 mitosesper 10 high-power field, absenceoflymphovascularinvasionandfreemarginswitha clear-ingdistanceof 1.5mm.Hematoxilyn-eosinstaining(Fig.1)and Immunohistochemical(IHC)analysisshowedpositivestainingfor carcinoembryonicantigen(CEA),cytokeratin(CK)5,7andepithelial membraneantigen(EMA).

Shehad apast medicalhistory of appendicitisin childhood, anxious-depressivesyndrome,osteoporosis,hiatalhernia,obesity andsmokedabout20cigarettesaday.NewYorkHeart Associa-tion(NYHA)scorewas1andAmericanSocietyofAnesthesiologists (ASA)scorewas1.Thepatienthadnoanorexiaandweightloss andtheexaminationdidnotrevealanyinguinallymphadenopathy. Laboratorytests,includingbloodcount,biochemicalinvestigations andserologicalviralmarkerswerenormal.Theelectrocardiogram

https://doi.org/10.1016/j.ijscr.2018.10.047

2210-2612/©2018TheAuthors.PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBYlicense(http://creativecommons.

S.Reinaetal./InternationalJournalofSurgeryCaseReports53(2018)196–199 197

Fig.1. Nestsofmonomorphiccuboidalporoidcellswithprominentnucleoli(hematoxylin&eosinstain).

showedsinusrhythmandthechestradiographshowednosigns ofpleuralorparenchymallesions.Aftermultidisciplinary discus-sionandbasedonthesub-optimalclearingmarginweperformed are-excisionofthepreviouswoundtoensurewidersafety mar-ginsofatleast20mmsimilarlytosurgicalstrategyforotherskin tumorsandinparticularmelanoma.Atthattimeitwasalsodecided toperformaSLNB;preoperativelymph-nodescintigraphyshowed thepresenceoftwosentinellymphnodesintheleftgrointhat wereexcisedduringSLNB.Recoveryfromsurgerywas unevent-fulandthepatientwasdischargedonthefirstpost-operativeday. Histopathologicalexaminationfoundnosignsofresidualor satel-liteneoplasiainthesurgicalsampleandthetworetrievedsentinel lymphnodeswerenegativeformetastaticdisease.

Patientisdiseasefree7monthsaftertheoperationand contin-uesfollow-up.

2.2. Case2

During August 2017, a 65year-old female was admitted to ourdepartmentwithhistologicalfindingofEPCoftherightleg. One month before, sheunderwent surgical excision of a cuta-neous lesion of the right leg. This lesion appeared brownish, exophytic,withulceratedsurface,moresuggestiveforasquamous cellcarcinomathananulceratednodularbasalcellcarcinoma.The histologicalexamination revealeda poroidneoplasm extending intothereticular dermiswitha thicknessof 5mm, 10 mitoses per10high-powerfield,absenceoflymphovascularinvasionand freemarginswithaclearingdistanceof2mm.Fig.2showsthe hematoxylin-eosinstainpictureofthelesion.

She had a past medical history of hysterectomy and bilat-eral salpingo-oophorectomy for uterine fibromatosis, kidney transplantationforseverechronicrenalfailure,highblood pres-sure,aneurysmaldilatationof therightcommoncarotid artery, hypercholesterolemia,hyperparathyroidismandpreviousinferior myocardialinfarction.Laboratorytests,includingbloodcount, bio-chemical investigations and serum viral markers were normal.

Aftermultidisciplinarydiscussionandbasedonthesub-optimal clearingmarginweperformedare-excisionofthepreviouswound toensurewidersafetymarginsofatleast20mm.Itwasalsodecided toperform a SLNB;the pre-operativelymph nodescintigraphy showedthepresence of two sentinellymphnodes in theright inguinalsite.Thepatientunderwentenlargementofthesurgical excisionuntil20mmoffree marginfromthepreviousexcision andSLNBofthetwolymphnodesidentifiedpreoperatively. Recov-eryfromsurgerywasuneventfulandthepatientwasdischarged fromhospital onthefirst post-operative day.Histopathological examinationfoundnosignsofresidualorsatelliteneoplasiainthe surgicalsampleandthetworetrievedsentinellymphnodeswere negativeformetastaticdisease.Patientisdiseasefree7months aftertheoperationandcontinuesfollow-up.

3. Discussion

EPCisarareneoplasmarisingfromtheintra-epidermalductal portionoftheeccrinesweatglandandrepresentsapproximately 0.005%ofallcasesofmalignantepithelialneoplasms[6–8,12].

Elderlypatientsarethemostaffected,withapeakincidence betweenthe6th_{and7thdecadeoflife.Althoughitdoesnotseem}

tohaveapredilectionforsexorrace,somestudiesindicateaslight prevalenceinwomen[7,9].TheexactetiologyofEPCisunclear. Someauthorssuggestedapossibleassociationwithradiation expo-sureandimmunosuppressionalthoughanexcessivesunexposure doesnotseem tobeasignificantriskfactor [4].EPCmayarise denovoorcandevelopfromapre-existingbenignlesion;some clinicalsigns,suchasspontaneousbleeding,suddengrowthand ulcerationinalongstandingstablelesionmustleadtothe suspi-cionofmalignantdegeneration[10].ClinicallyEPCcanbepresented asanerythematousorviolaceousnodule,papuleorplaquewith aninfiltrativeorerosivepattern.EPCusuallyarisesonthelower extremities(44%),followedbythetrunk(24%),head&neck(23%), upperextremities(11%),andrarelyinvolvesotherareas[3,5,11]. Microscopically, EPCis characterized by a cluster of anaplastic

198 S.Reinaetal./InternationalJournalofSurgeryCaseReports53(2018)196–199

Fig.2.Eccrineporocarcinomacomposedofbasaloidcellswithfocalinfiltrationintothedermis(hematoxylin&eosinstain).

cellswithnuclearhyperchromasiaandimportantmitoticactivity, extendingfromtheepidermistothedermis,surroundedbyductal lumen.Robinsonetal.[3]reportedspecifichistopathologicfeatures ofEPCwhichmaybepredictiveofalessfavorableoutcome. Thick-nessisthemainprognosticfactorsforEPC.Tumorsgreaterthan 7mminthickness,aninfiltratingfrontoftumorcells,thepresence oflymphovascularinvasion,andgreaterthan14mitosesper high-powerfieldwerenotedtobeassociatedwithapoorerprognosis [3].

Thedifferentialdiagnosisincludesbasalandsquamouscell car-cinoma,adenocarcinoma,amelanoticmelanoma,Bowen’sdisease, Paget’sdiseaseandalsobenignlesionslikefibromaandpyogenic granuloma.Someimmunohistochemical markersas carcinoem-bryonicantigen(CEA),EMA,andp53proteinmayplaya rolein thediagnosisofEPC[13].

Therapeutic options for the treatment of EPC include elec-trofulguration, electrocautery, surgical excision, radiation and amputation.Surgicalexcisionwithhistologicallyclearmarginsis generallyconsideredthetreatmentofchoicewithcureratesas highas70–80%,althougharecurrencerateofupto20%hasbeen reported[10].Thiselevateincidenceoflocalrecurrencemaybe duetoanotoptimalfreemarginatsurgicalexcision.Lymphnode metastasesarepresentatdiagnosisin20%ofcasesandthe inci-denceofvisceralmetastasesisreportedtobe10%[8,9].Thetumor tendstospreadtangentiallyinthelowerthirdoftheepidermis, thenafterinfiltratesthedermis,subcuticularfat andlymphatic system.Theroleofsentinellymphnodebiopsyremains contro-versial.Nourietalreportedaseriesofsixpatientsunderwentto SNLBwhichwerenegative[14].Sahnand Langinvestigatedthe roleofSNLBinhighriskEPC,theyreportednopositiveSLNB iden-tifiedamongsixpatientsbutonelocalandonedistantrecurrence onfollow-up[15].

BasedontheobservationthatEPCistumorwithanimportant lymphnodetropismsomeauthorshavesuggestedthepossiblerole ofSLNBforallorsomeEPCpatients,butmorestudiesareneeded [16].WeexploredSLNBapproachinthetwoobservedEPCpatients

withnonconclusiveresultsbecauseinbothofthemretrieved sen-tinelnodeswhereshowntobefreefrommetastasis.

4. Conclusions

EPCisararemalignancytumor;evenifitcanarisedenovo,it oftenoriginatesfromthetransformationofaporoma.The diag-nosisofEPCshouldbeconsideredinthedifferentialdiagnosisof skinlesions;earlydiagnosisisimportantbecauseinterferenceat anearly stagecouldprevent thehighrates of localrecurrence andmetastasis.Basedonitslowincidenceratesscantprotocolsor guidelinesareavailableforitsdiagnosisandmanagement.Inthe currenttwocasereports,weadoptedSLNBasastagingand diag-nostictoolsimilarlytoothermalignantneoplasmsoftheskinwith aconvergencebetweenthenon-pathologicalfindingofthelymph nodesandtheabsenceofdiseaseevenwithalimitedfollow-up. Currently,thestatusoftheSLNBaffectstheclinicalstageofpatients withEPCbutitsprognosticorpotentialtherapeuticroleremainto bedeterminedwithLargerstudiesandextendedfollow-up.

Conflictofinterest

None.

Sourcesoffunding

None.

Ethicalapproval

Thepaperisnotaresearchstudyandisnotrequireaethical approvalinourinstitute.

S.Reinaetal./InternationalJournalofSurgeryCaseReports53(2018)196–199 199 Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereport.

Authorcontribution

SimonaReina,ideatedthestudyanddraftedthearticle.

DenisePalombo,substantialcontributionstoconceptionand

design.

AlexandruBoscaneanu,acquisitionofdata.

NicolaSolari,revisingitcriticallyforimportantintellectual

con-tent.

SergioBertoglio,revisingitcriticallyforimportantintellectual

content.

LucaValle,acquisitionofdata.

FerdinandoCafiero,finalapprovaloftheversiontobe

submit-ted.

Allauthorsapprovedthefinaldraft.

Registrationofresearchstudies

OurresearchwasrecordedonClinicatrial.gov. TheclinicaltrialidentifiernumberisNCT03647631. OtherstudyIDnumber:Chirurgia1.

Guarantor

Dr.ssa Simona Reina. Department of Surgery, Chirurgia 1 OspedalePoliclinicoSanMartino,Padiglione15,IIIpianoponente -LargoRosannaBenzi,1016132–Genova,Italia.Tel.3492634555.

E-mail:simona.reina10@gmail.com.

Provenanceandpeerreview

Notcommissioned,externallypeerreviewed.

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