Use of a mixture of Lipiodol and Cyanoacrylate in percutaneous embolization treatment of symptomatic renal Angiomyolipomas: Our experience

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Case

Report

Use

of

a

mixture

of

Lipiodol

and

Cyanoacrylate

in

percutaneous

embolization

treatment

of

symptomatic

renal

Angiomyolipomas:

Our

experience

Simona

Caloggero,

MD

a

_,

_Francesca

_Catanzariti,

_MD

b

_,

_Alberto

_Stagno,

_MD

b

_,

Salvatore

Silipigni,

MD

b

_,

_Antonio

_Bottari,

_MD

b,∗

a_Department_of_Diagnostic_Imaging_and_{Radiotherapy,}_University_Hospital_“G._Martino”,_Messina,_Italy b_Department_of_Biomedical_and_Dental_Sciences_and_{Morphofunctional}_Imaging,_University_of_Messina,_Via_S. Camillo8,98122Messina,Italy

a r t i c l e

i n f

o

Articlehistory: Received4October2018 Revised17November2018 Accepted25November2018 Keywords: Angiomyolipoma Embolization

a b s

t r a c t

Purposeofthisreportistodescribesafetyandeffectivenessofselectiveartery emboliza-tioninthetreatmentofbleedingangiomyolipomas(AMLs)ofthekidneyusingamixtureof LipiodolandCyanoacrylate.

Two patients with bleedingAMLs underwent to superselective embolization of the lesionsusingmicrocatheterandLipiodolmixedwithCyanoacrylateintheratio3:1.

Primarybleedingcontrol ratewas100% withnomajorcomplications.Follow-upCT (meantime18months)demonstrateda significantreductioninsize(about50%)ofthe lesions.

Inconclusion,selectivearteryembolizationwithLipiodolandCyanoacrylateappearto besafeandeffectiveinthetreatmentofbleedingAMLs.

Introduction

Angiomyolipomas(AMLs)arebenignmesenchymaltumorsof thekidney,withanincidenceofabout0.3%-3%.Theyare com-posedofadiposetissue,abnormalvessels,andsmoothmuscle cells,invariousamounts.

Authorsdeclarenoconflictofinterest.

∗ _{Corresponding}_author.

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

Theycanbefoundassporadic(80%)orinassociationwith other disorders (eg, tuberous sclerosis, lymphangioleiomy-omatosis)[1].

Sporadic AMLs show preference for female gender (F:M=4:1) andtheyare usuallydiagnosedinasymptomatic patients during radiological examinations performed for

https://doi.org/10.1016/j.radcr.2018.11.019

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other reasons or in patients with nonspecific symptoms (eg,flankpain).

Commonlypatientswithtuberoussclerosishavemultiple, bilateral,andlargelesionswhichcaneasilybleed.Although mostofAMLsareasymptomatic,theycancausehematuria, abdominal,andretroperitonealbleeding,resultingin sponta-neouspain[2].

InasymptomaticpatientswithincidentalfindingofAML, thepredictivebleedingfactorsthatrequireapreventive treat-mentare:tumorsizemorethan4cm,presenceofintralesional aneurysmslargerthan5mm,andassociationwithtuberous sclerosis[3,4].

Treatment of AMLs includes radical nephrectomy, nephron-sparingtechniques(surgicalintervention, percuta-neous RF ablation, cryoablation,or microwave ablation),or percutaneousembolization,thelatteriscurrentlyconsidered thetreatmentofchoice[4,5].

Theaimofthispaperistoreportourexperienceinthe treatmentofbleedingAMLsofthekidneyusingamixtureof LipiodolandCyanoacrylate.

Case report 1

A 39-year-old woman presentedto the Emergency Depart-mentofourinstitutionwithsevereabdominalpain, hema-turia,andhypovolemicshock.

Afterpreliminaryexaminationoftheabdomenwithfast ultrasound, the patient underwent computed tomography angiography (CTA),which demonstrated the presenceof a high-attenuationfluidcollection(meanHU:47)surrounding therightkidneyduetoacutebleedingandmultiplebilateral renallesionscharacterizedbythepresenceoflowattenuation areasoffattissue(−20UHorless),typicalappearanceofAMLs. Oneofthem,locatedattheupperpole,hadanintralesional hyperdensespotcompatiblewithpseudoaneurysm(Fig.1).

Afterevaluationofrenalfunctionthrough blood labora-torytests(GFR,creatinine) and hemodynamicstabilization, endovasculartreatmentwasdecidedinordertoachieve se-lectivedevascularizationofthelesion.

Vascularaccesswasobtainedthroughfemoralapproach withanangiographiccatheter5Fr(Cordis,Fremont,CA); af-terselectivekidney angiography,superselective catheteriza-tionofthearterialbranchafferenttothetargetlesionswas performedwitha2.5Frmicrocatheter(Cook,Bloomington,IN)

[6].

Digitalsubtractionangiographywasobtainedtoevaluate thebloodflowoftheintralesionalarterialbranchesandthen it was decided to proceedwith embolization using a mix-tureofLipiodolandCyanoacrylate(Glubran,GEM,Italy)inthe ratio3:1.

Angiography controlafter embolizationmixture deploy-mentwasperformed(Fig.2).

Technicalsuccesswasreachedpreventingnontarget em-bolizationortheneedofreintervention.

Norintraorperiproceduralcomplicationswerereported. Renalfunctiontestswereperformedafter48hours.Patient wasdischargedafter10dayswithoutrenaldysfunction.

Fig. 1 – Case report 1. Coronal CTA reconstruction. (a) Arterial phase. High-attenuation (mean HU: 47.3) fluid collection surrounding the right kidney due to acute bleeding (circle). At the upper pole is visible a hyperdense spot compatible with intralesional aneurysm (white arrow). (b) Delayed phase. The bleeding lesion is well-defined in close relationship with hematoma at upper pole (white arrowhead). Another AML, with a considerable amount of fat tissue, is visible at the upper pole of the left kidney (black arrows).

AML, Angiomyolipomas.

No moreepisodes ofbleedingwere reported duringthe observationtime.

Case report 2

A21-year-oldmanaffectedbytuberoussclerosiswasreferred toourDepartmentwithflankpain.

ThepatientunderwentMRIwhichdemonstratedthe pres-enceofmultipleandbilateralrenallesions,withsizeranging from10to50mmandtypicalappearanceforAMLs.

Oneofthemcausedcompressiononipsilateralpsoas mus-cle.

According withthe patientandafterevaluationofrenal functionwasdecidedtoembolizethelesion.

PreliminaryCTAclearlyshowedarterialvascularizationof thekidneyandofthetargetlesion(Fig.3).

Theprocedure was performedwiththe same technique and materials of the previous case and was technically successful.

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Fig. 2 – Case report 1. (a) Selective renal angiography revealing active bleeding at the upper lobe of the right kidney (white arrowhead). (b) Superselective angiography from the feeding artery. (c) Single control showing Lipiodol and Cyanoacrilate distribution (white arrowhead). (d) Final

DSA demonstrating complete devascularization of the AML

and normal angiographic pattern of the surrounding renal parenchyma.

AML, Angiomyolipomas; DSA, digital subtraction angiography.

Thepatient developedpostembolization syndrome with nausea,flankpain,and vomitaftertheprocedure,however postprocedureCTscandidnotshowedanycomplication.

Postembolizationsyndromewassuccessfullytreatedwith intravenous administration of analgesics, antibiotics, and antiemetic.

Patientshad ahospitalization time of2 daysand renal functiontestswereperformedafter48hours.Noneofthe pa-tientshadrenaldysfunction.

Atfollow-upCTscanasignificantreductioninsize(mean 50%)ofthetreatedlesionswasreported,whichthenremained stableduringseveralcontrols(Fig.4).

Discussion

AMLsarethemostcommonbenignkidneytumors,composed ofabnormalbloodvessels,smoothmuscle,andadiposetissue. Spontaneousbleeding,theirmaincomplication,iscausedby thelow-elastincontent inthe vesselwallsthatcanleadto aneurysmaldegeneration.Thebleedingisparticularly com-moninlargelesions(morethan4cm);inthesecasesa pre-ventivetreatmentisoftennecessary[7].

Several treatment options are available: nephrectomy, nephronsparingtechniques,orpercutaneousembolization.

Fig. 3 – Case report 2. Maximum intensity projection (MIP) reconstruction showing a small artery (white arrow) feeding the target lesion which compressed psoas muscle (black arrowheads).

Nowadaysthelatteroptionisassumingasignificantrole inthetreatmentofAMLsbecauseitsminimally-invasiveness andguaranteesaquickapproachinacutebleedingandallows agoodprophylaxisforlargelesions.

Moreover, in patients with bilateral involvement of the kidneys,thankstothesuperselectivecatheterizationofthe vascularbranchesafferenttothe lesions,itoffersthegreat chancetospareasmuchnormalrenalparenchymaas possi-ble[8].

Indeedthe2patientsreportedinthispaperhadbilateral le-sions,thereforetotalorpartialnephrectomywasnotthebest optionstopreserveanoptimalrenalfunction.

Ahigh-qualitypercutaneousembolizationrequiresa care-fulchoiceofembolicmaterials,whichismainlybasedon op-erator’sexperience,consideredthelackofcomparative stud-iesbetweendifferentembolicagentsforAMLstreatment[4,9]. Althoughmanyembolicmaterialsarenowadaysavailable, themainonesarepolyvinylalcohol(PVA),absolutealcohol andspirals[1,9].

Recently some preliminary experiences with ethylene vinylalcohol(EVOH)copolymer(Onyx;Medtronic, Minneapo-lis,MN)werealsoreportedwithexcellentsuccessrate[5].

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Fig. 4 – Case report 2. (a, b) Axial and coronal CTA images before embolization showing a low-attenuation mass in the right kidney which compresses the psoas muscle causing flank pain (white arrows). (c, d) Axial and coronal CT scans 3 months after treatment demonstrating significant shrinkage of the AML with no more muscular compression (black arrows).

However,evenLipiodol(Guerbet,Villepinte,France), com-monly used inthe treatment of hepatocellular carcinoma, may have a role as an embolic agent for the renal AMLs, thoughsuchuseisstillnotsocommonuptonow[10].

Eachembolic materialhasits ownadvantages and dis-advantages.Someauthorshaveshownthattheparticlesof PVAcannotpassthroughthecapillarywallswithinthelesion, causingonlypartialocclusion,whereastheuseofspiralscan leadtotheformationofcollateralvascularbranches. Further-morethesearehigh-costmaterials [11,12].

Tothebestofourknowledgenocaseshavebeendescribed inliteraturetreatedwithCyanoacrylateandLipiodol.

Inourexperiencetheuseofthismixturehadshowna sat-isfactorycapacityofdiffusionandperfusionresultinginthe interruptionofthebloodflowwithintheAMLs.

Unlike others embolizing materials, Lipiodol is a ra-diopaque contrast medium, detectable under fluoroscopy, providingabetterawarenessoftheproceduretothe radiolo-gist [13].

Theuseofsystemsofmicrocathetersandthepossibility ofobservingthemixtureofLipiodolandCyanoacrylateunder fluoroscopymakepossibletheachievementofthetarget vas-culardistricts(withagreatpercentageofsparedrenaltissue) andpermittoidentifyanyrefluxthatmightaffectthefinal successoftheprocedure.

InadditionLipiodolshowedits usefulnessinthe follow-upofthepatients,helpinginidentifyingthetreatedlesions becauseofitshighaffinityforthetumorvesselsanditslong persistence,associatedwithlowerstreakartifactthanother radiopaqueembolizingmaterials(eg,coilsorEVOH)[5](Fig.5). In our experience, percutaneous embolization of renal AMLwithamixtureofCyanoacrylateandLipiodolhasproved tobeasafeandeffectivemethodinthetreatmentof

symp-Fig. 5 – Case report 2. Axial MIP. Presence of radiopaque material 3 months after embolization demonstrating long persistence of Lipiodol inside the lesion (black arrows). Absence of any significant streak artifact allows good evaluation of AML enhancement (also perceptible in Fig.4c and d).

tomaticpatients,evencritical,preservingthe healthyrenal parenchyma.

Thisisextremelyimportantforcriticalpatientswith mul-tiplelesions,oftenlargeandbilateral,whereinsurgerywould betoodemolitive.

In particular, the embolization with the mixture of CyanoacrylateandLipiodol, injectedbyexperienced opera-tors,allowsrapidembolizationofthevesselswithdecreased operatingtimeandradiationdose tothe patientandlower costscomparedtootherembolizationmaterials(spirals,PVA, EVOH).

In conclusion,CyanoacrylateandLipiodol couldbe con-sideredasanotherembolicoptionforthetreatmentofAML, notonlyforimmediatebleedingcontrolbutalsoforlong-term preventionofrebleeding.

R E F E R E N C E S

[1] VillaltaJD, SorensenMD, DurackJC, KerlanRK, StollerML. Selectivearterialembolizationofangiomyolipomas:a comparisonofsmallerandlargerembolicagents.JUrol 2011;186:921–7.

[2] AndersenPE, ThorlundMG, WennevikGE, PedersenRL, LundL.Interventionaltreatmentofrenalangiomyolipoma: immediateresultsandclinicalandradiologicalfollow-upof 4.5years.ActaRadiologicaOpen2015;4(7):1–8.

[3]HocqueletA,CornelisF,LeBrasY,MeyerM,TricaudE, LasserreAS,etal.Long-termresultsofpreventive embolizationofrenalangiomyolipomas:evaluationof predictivefactorofvolumedecrease.EurRadiol2014. doi:10.1007/s00330-014-3244-4.

[4] FlumAS, HamouiN, SaidMA, YangXJ, CasalinoDD, McGuireBB, etal. Updateonthediagnosisandmanagement ofrenalangiomyolipoma.JUrol2016;195:834–46.

(5)

[5] UrbanoJ, PaulL, CabreraM, Alonso-BurgosA, GomezD. Electiveandemergencyrenalangiomyolipomaembolization withethylenevinylalcoholcopolymer:feasibilityandinitial experience.JVascIntervRadiol2017;28:832–9.

[6] LiD, PuaBB, MadoffDC.Roleofembolizationinthe treatmentofrenalmasses.SeminInterventRadiol 2014;31:70–81.

[7] BishayVL, CrinoPB, WeinAJ, MalkowiczSB, TrerotolaSO, SoulenMC, etal. Embolizationofgiantrenal

angiomyolipomas:techniqueandresults.JVascInterv Radiol2010;21:67–72.

[8] ChanCK, YuS, YipS, LeeP.Theefficacy,safetyanddurability ofselectiverenalarterialembolizationintreating

symptomaticandasymptomaticrenalangiomyolipoma. Urology2011;77:642–8.

[9] MurrayTE, DoyleF, LeeM.Transarterialembolizationof angiomyolipoma:asystematicreview.JUrol2015;194:1–5.

[10]HuangQ,ZhaiRY.Embolizationofsymptomaticrenal angiomyolipomawithamixtureoflipiodolandPVA,a mid-termresult.ChinJCancerRes2014;26(4):399–403 PMCID:PMC4153937.doi:10.3978/j.issn.1000-9604.2014.07.04.

[11] TakebayashiS, HorikawaA, AraiM, IsoS, NoguchiK. Transarterialethanolablationforsporadicand non-hemorrhagingangiomyolipomainthekidney.EurJ Radiol2009;72(1):139–45.

[12] ChatziioannouA, GargasD, MalagariK, KornezosI, IoannidisI, PrimetisE, etal. Transcatheterarterial embolizationastherapyofrenalangiomyolipomas:the evolutionin15yearsofexperience.EurJRadiol 2012;81:2308–12.

[13]ChickCM,TanBS,ChengC,TanejaR,LoR,TanYH,etal. Long-termfollow-upofthetreatmentofrenal

angiomyolipomasafterselectivearterialembolizationwith alcohol.EurRadiol2014.doi:10.1007/s00330-014-3244-4.