Case report of a successful multidisciplinary approach to a giant scrotal malformation

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InternationalJournalofSurgeryCaseReports69(2020)24–27

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

report

of

a

successful

multidisciplinary

approach

to

a

giant

scrotal

malformation

A.

Ascoli

Marchetti

_,

_G.

_Citoni

_,

_R.

_Gandini

_,

_A.

_Ippoliti

a_{VascularSurgeryUnitBiomedicineDepartment,UniversityofRomeTorVergata,Italy} b_{RadiologyUnit,UniversityofRomeTorVergata,Italy}

a

r

t

i

c

l

e

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n

f

o

Articlehistory:

Received12November2019 Receivedinrevisedform8March2020 Accepted13March2020

Availableonline28March2020 Keywords:

Selectiveangiography Embolization

Giantarteriovenousmalformation Previoussurgery

Multidisciplinary

a

b

s

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r

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t

INTRODUCTION:Congenitalpelvicmalformationsarerareandrepresentadifficulttherapeuticchallenge. Scrotalarteriovenousmalformationsarequiteunusual,withonlyafewsuchcasesreportedinthe liter-ature.Onlyonecaseofscrotalmalformation?duetoanarteriovenousfistularesultinginazoospermia hasbeendescribed.

PRESENTATIONOFCASE: Thetwo-phasestrategyadoptedinthatcasepermittedcompletetreatmentof alarge-sizedmalformationthatwasservedby4mainbloodconfluences.

DISCUSSION:Angio-CTperformedusingreconstructionswithMIPandSSDalgorithmsprovidedmore detaileddataabouttheextensionoftheafferentsandefferentsofthearteriovenousmalformation,thus enablingustoplantheendovasculartreatmentofthelesion.

CONCLUSION:Amultidisciplinaryapproachismandatorytoachievegoodresultsinthesecases. ©2020TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen

accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

1. Introduction

Thispaperis being reportedin linewiththeSCARE criteria [1].Congenitalpelvicmalformationsarerareandrepresenta dif-ficulttherapeuticchallenge.Scrotalarteriovenousmalformations arequiteunusual,andonlyafewsuchcaseshavebeendescribed intheliterature.Duetotheircomplexpathologyandalackof avail-abledata,thediagnosisandtreatmentofthistypeofcasesrequire amultidisciplinaryapproachtotreatthelesionandpreserveboth thefertilityandthesexualfunctionalityofthepatient.

2. Clinicalcase

AttheVascularSurgeryDepartmentofourInstitution,we exam-inedamalepatient(42yearsold)whopresentedwitha scrotal tumefaction.This tumefaction was approximately 15 × 17 cm insizeand had beenprogressivelydeveloping aftersurgery for theremoval of a left-side testicularangioma. Secondary steril-ityhad occurred, assuggested bya spermiogramthat revealed azoospermia (<20,000 spermatozoa/mL). Ultrasonographic test resultsperformedduringhospitalizationrevealedadiagnosisof scrotalarteriovenousmalformationandasmallfluidslopeinthe lefttunicavaginalis;thetesticlesappearedtoberegularinshape andhadanechographicpattern.Thepatientunderwentan

angio-∗ Correspondingauthorat:VascularSurgery,BiomedicineandPrevention Depart-ment,PoliclinicoTorVergata,VialeOxford81,00133Rome,Italy.

E-mailaddress:[email protected](A.AscoliMarchetti).

computedtomography(CT)ofspiralmultislidesoftheabdominal aortaandthelowerlimbs.Theexamresultsindicatedthe pres-enceofa rawagglomerate of enlargedbloodvessels locatedin theleft-1sidescrotalportionthatwascausingacontralateral dis-locationof therighttesticle (Fig.1).The masswassuppliedby fourarterialconfluences:twofromthesuperficialfemoralarteries andtwofromthehypogastricarteriesthroughthebilateralpenile arteries.In addition,thescrotalvenoussystemshowed varicos-ityandcongestionof thebilateralcommonfemoralveins,most likelyduetoanarteriovenousshunt.Itwasdecidedthatthe mal-formationshouldbetreatedonthebasisofaprotocolinvolving endovasculararterialembolization withGlubran 2.Thepatient, aftersigninghisinformedconsent,underwentaselective catheter-ization ofthearterialramifications viaa left-sidepercutaneous transfemoralapproachoriginatingfromtheleftfemoralandleft hypogastric arteries.A postproceduralangiographic check indi-cateddevascularizationofthelesionandaslightreductioninthe sizeofthescrotaltumefaction.Twodaysaftertheprocedure,a sec-ondembolizationwasperformedviaalefttransfemoralapproach toobtainafurtherreductioninthevascularizationofthe tume-faction,combinedwiththeselectivecatheterizationofthearterial ramificationscomingfromtherightfemoralandrighthypogastric arteries(Fig.2).After72hfromtheembolizationaskinnecrosison thehemiscrotumduetoischemiaoftheareapreviouslysupplied bytheanomalousbloodvesselwaspresent.Toavoidthe devel-opmentofaseriousformofinfectednecrosis,suchasFournier’s gangrene,thepatientunderwentrepeatsurgeryanddebridement of thewide necrotic skintissue (Fig.3A B).During the follow-ingdays,thepatientreceiveddailymedicationsatthelesionsite,

https://doi.org/10.1016/j.ijscr.2020.03.019

2210-2612/©2020TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBYlicense(http://creativecommons.

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A.AscoliMarchettietal./InternationalJournalofSurgeryCaseReports69(2020)24–27 25

Fig.1.Angio-CTMIPofthelowerabdomenandthecranialportionofinferiorlimbs:Seethepresenceofarawagglomerateofenlargedbloodvesselslocatedintheleft-side ofthescrotumandcontralateraldislocationoftherighttesticle.

Fig.2. Angio-CTSSDreconstructionswowingnormalvasculararisationoftheiliofemoralareaandtheselectiveembolisationwithGluedepositionintheA-Vmalformation andinthearterialramificationscomingfromtherightfemoralartery.

coupledwithendovenousantibiotictherapy.Theagglomerate pro-gressivelydecreased insize,and thenecrotic areaincreasedits demarcation.Thepatientwasdischargedfromthehospitalwith antibiotictherapy20daysafterthefirstembolizationprocedure. Inaddition,hewasprescribeddailymedications,andhe under-went subsequentplastic surgery for scrotal reconstruction.The spermiogramresultshowedazoospermiainbothtesticles.

3. Discussion

Testicular arteriovenous malformations arevery rare lesions thatrepresentlessthan 1%ofvascular neoformations;theycan becongenital,traumaticoriatrogenous[2–4].Becauseofthe rar-ityofthispathology,itisnotalwayseasytoobtainanaccurate diagnosisorpropertreatment.Typically, thetreatmentincludes

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Fig.3. A.Righthemiscrotumaspectafternecrotictissueasportation.B.Necroticskintissueremoved.C.Sixmontsappearanceaftertheprocedure.Notethevolumereduction ofthescrotum.

surgicalknotsortheembolizationandsuccessiveremovalofthe agglomerate[5].Inthecasedescribedhere,thepatient’scondition wascomplicatedbyapreviousoperationthathadbeenperformed toremoveascrotalangioma.Afterthatsurgery,aprogressiveand significantincreaseinthesizeofthelesionwasobserveduntilit reachedalargesizeandimpactedthepatient’sfertility[6].Thus, itisfundamentaltoobtainacorrectandpreciseevaluationofthe natureandtypeofvascularizationofthistypeoflesioninorder tointerveneappropriately,thuspreventingrecurrenceand pre-serving,asmuch aspossible,thehealthy surroundingtissueso that deleterious effects in the sexualand reproductive spheres canbeavoided.Angio-CTperformedusingreconstructionswith MIPandSSDalgorithmsprovided moredetailed dataaboutthe extensionoftheafferentsandefferentsofthearteriovenous mal-formation,thusenabling ustoplan theendovasculartreatment ofthelesion.Duetothelargesizeand positionofthe agglom-erate,itwasdecidedthattheembolizationwouldbecompleted intwophasessoastominimizetherisksandischemic complica-tionsentailedintheoperation,suchaspossibleadditionalfertility damage,oerigendiimpotenceandthepossiblerecurrenceofa fis-tula[7,8].Theangiographicexaminationmadeitpossibletoobtain amoredetailedevaluationofthelesion’sanatomical configura-tionandtochoosethebesttherapeuticstrategytoemploy[9].The adoptedtwophasestrategypermittedcompletetreatmentofthe malformation,whichwaslarge-sizedandwasservedby4main bloodconfluences.Theembolizationcausedaprogressive reduc-tionintheagglomerate,andthis allowedsurgicalexcisionwith aminimum riskof bleeding, aswellasthesurgicalremoval of thenecrotic scrotalskinarea. Moreover,it waspossibleto pre-serve theintegrity of the didymuslocated nearthe lesion and avoidthedevelopmentofFournier’s gangrene.Theagglomerate decreasedinsizeuntil nearcomplete restoration(“restitutioad integrum”)wasachieved.Alaterreconstructiveplasticsurgery pro-cedurewasperformedtotreattheentireareaaffectedbythelesion [10](Fig.3C).

4. Conclusions

Scrotal malformations are quite rare, and they can involve problemsrelatedtothetherapeuticstrategy chosen.The multi-disciplinaryapproach usedin this case involveda less invasive treatmentprotocolandsubsequentlylessriskysurgery.This strat-egyallowedthecompleteexclusionofthemalformationandthe retentionoferectilecapacitybutdidnotimproveorganfunction, agoalthatremainedunmet.Toobtainaconclusiveevaluation,an additionalperiodoftimeisrequiredtomonitortheregressionof thetumefactioninthelongtermaswellastheaestheticresultsand eventualbenefitsregardingfertility.

Funding

No funding was declared and the acknowledgement are reportedattheendofthetext.Authorsdeclarethatthesponsor havenoroleinthestudy,inthecollectionoranalysisand interpre-tationofdata;inthewritingofthemanuscript;andinthedecision tosubmitthemanuscriptforpublication.

Ethicalapproval

The patient give the consensus to the procedure. Ethical approvalisexemptforcasereportsbyourinstitution.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonrequest.

Authorcontribution

Studyconceptordesig:AAM,GC,RGandAI. Datacollection:AAM,GC,RGandAI.

Dataanalysisorinterpretation:AAM,GC,RGandAI. Writingthepaper:AAM,GC,RGandAI.

Registrationofresearchstudies

ThispublicationisfreefromRegistrationofResearchStudies, becauseisnotusedanewtechnology.

Guarantor

Prof.ArnaldoIppoliti,Prof.AndreaAscoliMarchetti.

Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed.

DeclarationofCompetingInterest

Theauthorsdeclaretohavenoconflictsofinterestorany finan-cialandpersonalrelationshipwithotherpeopleororganisations thatcouldinappropriatelyinfluence(bias)ourwork.

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Acknowledgment

TheauthorsthankProf. GuidoVirgilioftheUrologyUnitfor collaborating in clinical practice and during the writing of the manuscript.

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