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InternationalJournalofSurgeryCaseReports46(2018)31–33
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International
Journal
of
Surgery
Case
Reports
j o u r n a l h o m e p a g e :w w w . c a s e r e p o r t s . c o m
Breast
cancer
cutaneous
metastases
mimicking
Papilloma
Cutis
Lymphostatica.
Biopsy
to
avoid
pitfalls
Giuseppe
Giudice,
Michelangelo
Vestita,
Fabio
Robusto,
Paolo
Annoscia,
Francesco
Ciancio
∗,
Eleonora
Nacchiero
DepartmentofPlasticandReconstructiveSurgery,UniversityofBari,Italy
a
r
t
i
c
l
e
i
n
f
o
Articlehistory: Received8January2018 Accepted19March2018 Availableonline10April2018
Keywords: Breastcancer Lymphedema
PapillomaCutisLymphostatica Metastases
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s
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r
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INTRODUCTION:Secondarylymphedemaisthemostfrequentlong-termcomplicationofaxillary lym-phadenectomy.Itcanresultincomplicationaserysipelas,warts,PapillomaCutisLymphostatica(PCL),or angiosarcomas.Moreover,inwomenaffectedbybreastcanceranaccuratedifferentialdiagnosisamong theseconditionsorcomplicationrelatedtoradiationdermatitisorcutaneousmetastasisisessential. PRESENTATIONOFCASE:Wereportthecaseofa60-year-oldpostmenopausalCaucasianwomanaffected bysecondarylymphedemafollowingcompletemastectomyforbreastcancer.Thepatientaftersurgery wastreatedwithradiotherapy,chemotherapyandhormonetherapy,developingalympedemaofleft armafterfewmonths.TheselesionshadclinicaltypicalfeaturesofPCL,buthistopathologicalanalysis revealeddermo-hypodermicmetastasisofbreastcarcinoma.
DISCUSSION:Thepresenceofskinlesionsinsecondarylymphedemaafteroncologicallymphadenectomy requiresanaccuratedifferentialdiagnosis.Infact,theselesionscanemulatedegenerativeorinfective skindiseases;anyway,inpatientsaffectedbysecondarylymphedemaotherlesscommonconditions– asPLC,nodular-typelichenmyxedematosusorGottron’scarcinoidpapillomatosis–shouldbetakeninto account.
CONCLUSION:Ourcasereportsthepossibilitythatmetastasesofbreastcancermightalsomimicthese conditions.
©2018TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).
1. Introduction
Secondarylymphedema ofupper limbsisthemostfrequent long-term complication of axillary lymphatic tissue resections, withanincidencerangingbetween6%and30%[1].Mostfrequent sequelaoflymphedemaareerysipelasandwarts,butinveryrare casesit canresultin moreseverecomplications, suchas Papil-lomatosisCutis Lymphostatica(PCL)or angiosarcomas.PCLis a benign,usuallyasymptomaticandunderreportedcondition result-ingfromchroniclymphedema[2].Itwasmorefrequentlyreported inprimarylymphedemaorascomplicationofasecondary dam-ageoflymphaticvesselsduetodiabetes.Todate,fewcasesofPCL havebeendocumentedafteraiatrogenicsecondarylymphedema [3,4].Alsocarcinomasorangiosarcomasarisinginlimbsaffected bychroniclymphedemahavebeendocumented[5,6].Moreover, lesionsarisingin upper limbin women affectedby breast car-cinoma could be related to cutaneous metastases or radiation dermatitisfollowingradiationtherapy[7,8].Forthis reason,the
∗ Correspondingauthorat:DepartmentofPlasticandReconstructiveSurgery, UniversityofBari,CAP70124PiazzaGiulioCesare11,Bari,Italy.
E-mailaddress:francescociancio01@gmail.com(F.Ciancio).
presence ofskinlesionsoftheupperlimbsin patientsaffected bybreastcancerrequiresanaccuratedifferentialdiagnosisamong cancerrecurrencesormetastasis,therapeuticalcomplications,and dermatologicalconditions.Theworkhasbeenreportedinlinewith theSCAREcriteria[9].
2. Casereport
Wepresentthecaseofa60-year-oldpostmenopausalCaucasian woman affectedbysecondary lymphedema following complete mastectomyandaxillarydissectionforaLuminalBinvasivelobular carcinomaofleftbreast.Immunohistochemistrytestifiesnegativity forHER-2mutation.Patientunderwentmastectomyandcomplete lymphadenectomyofleftaxillaryregioninNovember2015.The patientrefusedthereconstructivesurgicaltreatmentofthebreast. After surgery, the case was presented to the multidisciplinary breastcancerboardofourinstitutionandthepatientunderwent localradiotherapyandadjuvantchemotherapybasedon anthracy-cline/taxaneregimenfollowedbyhormonetherapywithletrozole. InOctober2016,thepatientpresentedwithastageIIIleftupper limblymphedema[10]withtheappearanceofisochromicnodular lesionsoftheskin(Figs.1and2).Segmentallymphoscintigraphy ofupperlimbsdocumentedaseverelymphedemaoftheleftupper
https://doi.org/10.1016/j.ijscr.2018.03.025
2210-2612/©2018TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://
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32 G.Giudiceetal./InternationalJournalofSurgeryCaseReports46(2018)31–33
Fig.1.Initialclinicalpresentationoflesions.
Fig.2. Detailofinitialnodularlesion.
Fig.3. Clinicalpresentationofmultiplenodularreddenedlesions.
limbwithahighvalueofTransportIndex(28),while lymphoscinti-graficexaminationwasnormal in thecontralateral upperlimb. Thepatientunderwenttherapywithphysiotherapy,bandagesand plasticdevices;inDecember2016,however,thepatientreported furtherdiffusionandreddeningof theskinlesionsaffectingthe leftupperlimb(Figs.3and4).Thephysicalexaminationrevealed thepresenceofpainlessnon-confluentovalnodularlesions, dis-tributedonthevolarsurfaceofleftarmand forearm.Clinically, theselesionshadthetypicalfeaturesoflymphostatic papillomato-sis;nonetheless, due tothepatient’s history, we performedan excisionalbiopsyofone ofthelesions.Histopathologicanalysis revealedthepresenceof0.7cm masscharacterizedbypresence ofpleomorphicspindlecellswithlargehyper-chromaticnucleiand denseeosinophiliccytoplasm.Immunohistochemistrywaspositive forproteinKi67andestrogenreceptor,confirmingthediagnosisof dermo-hypodermicmetastasisofinvasivebreastcarcinoma.The patientwasconsideredhormone-refractory and sheunderwent tofirstlinechemotherapywithanti-VEGFincombinationto
tax-Fig.4.Detailofreddenedlesions.
ane.Todate,shehasundergoneclinicalandimaging(chestX-ray, abdominalultrasound,andwhole-bodySPECT/CTscan)follow-up withnoevidenceoflymphaticorvisceralmetastases.
3. Discussion
Presenceofovalexophiticnon-pigmentedlesioninthe homo-lateralupper limb of womanwho underwent mastectomy and completelymphadenectomyofaxillaryregionforabreastcancer needanaccuratedifferentialdiagnosis,evenmoreinpresenceof lymphedema.
First of all,differential diagnosis includes degenerative skin conditions such as keratoacanthoma, pilomatricoma, pyoderma vegetans, and non-pigmented skin cancers; however the pres-enceofmultipleexophiticlesionsmakestheseunlikelydiagnoses. Infectivecomplicationsoflymphedemaaremoreplausibleinthis patient;infact,lymphedemaisthemostcommonlong-term com-plicationofaxillarylymphnodedissection[1]andassociationof reducedimmunesurveillanceandskindamageinthe lymphede-matousregions[11]mayleadtoskininfections.Inmoresevere cases,lymphedemacanresultinpapillomatosiscutis lymphostat-ica(PCL),characterizedbythepresenceofmultipleskin-colored, confluent,partlyhyperkeratotic,verrucouspapulesonlimbs[6].In suchcases,skininfections,aserysipelas,mayresultfromskinlossof continuity.Wartsandangiosarcomaswerealsoreportedinpatients affectedbyPCL[5,6]. Finally,nodular-type lichen myxedemato-susandGottron’s carcinoidpapillomatosis[12]skinshouldalso beconsideredincaseofmultipleexophyticlesionsonlimbs,but theabsenceofhypothyroidismandthenegativityforHPVserology assayexcludethesediagnoses.
Alloftheaboveconditionsarecompatibleswiththeskinlesions affectingtheleftupperlimbofourpatient;cutaneousmetastasesof breastcancer,ontheotherhand,havebeendocumentedinseveral studies,asevidencedbyourresearchatoneofthemost impor-tantdatabasesintheliterature[13].Thesecanbepolymorphous, mimickingwhealrash[14],erythemaannullarecentrifugum[15], nodules[16]orexophyticlesions[7].
Inanycase,excisionalbiopsyisindispensableforacorrectand precociousdiagnosisofskinmetastasisofbreastcancermimicking PCLinalimbaffectedbylymphedema.Furthermore,asinourcase, skinmetastasesmaybethefirstclinicalevidenceofrecurrenceand theirdetectionimpliestheadjustmentofchemotherapyregimen andthusholdsprognosticandtherapeuticsignificance.
Conflictsofinterest
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Funding
Authorshavenotreceivedfundingorsponsorforpaper produc-tion.
Ethicalapproval
Tocarryoutthisscientificwork,therewasnoneedtoresort totheethicscommittee.Ethicalapprovalhasbeenexemptedfrom ourinstitution.
Consent
Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonreques.
Authorcontribution
Alltheauthorscontributedequallytothewritingofthepaper. Researchregistrationnumber
NA. Guarantor
FrancescoCiancioMD. References
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