MR imaging of paratesticular bilateral leiomyoma: A case report

(1)

R a d i o l o g y C a s e R e p o r t s 1 4 ( 2 0 1 9 ) 5 9 1 – 5 9 4

Available

online

at

www.sciencedirect.com

journal

homepage:

www.elsevier.com/locate/radcr

Case

Report

MR

imaging

of

paratesticular

bilateral

leiomyoma:

A

case

report

Serena

Dell’Aversana,

MD,

Arnaldo

Stanzione,

MD

∗

,

Valeria

Romeo,

MD,

Marcello

Caggiano,

MD,

Pietro

Gisonni,

MD,

Luigi

Insabato,

MD,

PhD,

Simone

Maurea,

MD,

PhD

DepartmentofAdvancedBiomedicalSciences,UniversityofNaplesFedericoII,Italy

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received12January2019 Revised20February2019 Accepted23February2019 Keywords: Paratesticular Leiomyoma US MRI Diagnostics

a

b

s

t

r

a

c

t

Paratesticularleiomyomaisararebenignneoplasmthatmayarisefromsmoothmuscle cellscontainedineithertheepididymis,thespermaticcord,orthetunicaalbuginea. Usu-allypatientspresentapalpable,asymptomaticmass,withahigherprevalenceamongthe fourthandfifthdecadeoflife.Inthiscasereportwedescribea57-year-oldmanwith bilat-eralscrotalpalpablemassesevaluatedwithultrasoundandmagneticresonanceimaging thatweresuggestiveforleiomyoma.Thelesionsweresurgicallyremovedandpathology revealednosignsofmalignancyconfirmingthediagnostichypothesisofleiomyoma. Ultra-soundisconsideredtheimagingmodalityofchoicefortheinitialevaluationoftesticular massessinceitallowsanaccuratelocalization(ietesticularvsparatesticular)andcan iden-tifysignsofmalignancy.Magneticresonanceimagingislessfrequentlyperformedbutcan considerablyimprovelesioncharacterization.

Case

report

A57-year-oldmancametoourinstitutionforthediagnostic evaluationofbilateral scrotal palpablemasses.Thepatient was in good health, without medical records of note and completelyasymptomatic.Atesticular ultrasound(US)was performed,revealingthepresenceof2paratesticularlesions, corresponding to the palpable masses, measuring respec-tively45mmontherightsideand55mmontheleftside.The

Conflictofintereststatement:Theauthorsreportnoconflictofinterest.

Funding:Thisstudyreceivednofunding.

Consent:Informedconsentwasobtainedfromthepatientinvolvedinthispublication.

∗ _{Corresponding}_author.

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

lesionsappearedround-shaped,welldefined,andwith regu-larmargins,adherenttotheinternalspermfascia;theUSalso demonstratedahighlyheterogeneoustissuewithnosignsof vascularity onColor-Doppler (Fig.1).When blood testsand urine analysiswere performed,resultswere withinnormal ranges. Additionally, tumoral markers were researched (ie alpha-fetoprotein,beta humanchorionicgonadotropin, and antigen carcinoembrionar) with none found significantly altered.

https://doi.org/10.1016/j.radcr.2019.02.019

(2)

592

Fig.1– USdemonstrates2highlyheterogeneousparatesticularmasses,measuring45mmontherightside(A)and55mm ontheleftside(B).Thesemassesappearedround-shapedandwithregularmargins(C).ThetesticularUSpatterndidnot showsignificantanomalies(D).

Inordertofurthercharacterizethetesticularmasses,an MRI scan was performed. MR images confirmed the pres-enceinthescrotalsacofthe2heterogeneousparatesticular masses,1foreachside,noncapsulatedwithregularmargins. Themasses were hypointenseon T2-weighted images and isointenseonT1-weightedimages,withheterogeneous con-trastenhancementinthepostcontrastographicphase; more-over,theyweretightlyadherenttothespermaticcordand dis-placedthetesticles,whichpresentedregularsignalintensity (Fig.2).

The lesions were surgically removed and pathology re-vealednosignsofmalignancy.Inparticular,grossspecimen presentedmacroscopicallywhitishappearance(Fig.3).On mi-croscopicanalysis,amesenchymallesionwasfound,without cytologicalatypia,consisting ofbundlesofcells withfused nucleiandabundanteosinophiliccytoplasm; immunohisto-chemistryshowedastrongpositivityformusclespecificactin andfordesmin.Basedonthesefindings,thefinaldiagnosis wasofleiomyoma.Todate,thepatientisingoodhealthand hasnotsufferedfromcomplicationsaftersurgery.

Discussion

Paratesticulartumorsarerarebutclinicallysignificantlesions thataffectpatientsofallages; theyare mostlikelybenign,

withtheprevalenceofmalignanciesbeingapproximately3%

[1–3]. The majority of scrotal benign paratesticular lesions includelipoma, adenomatoid tumors,leiomyoma, fibroma, hemangioma,neurofibroma,and papillarycystadenoma[2]. Ontheother hand,malignant tumorsincludeliposarcoma, rhabdomyosarcoma, lymphoma, fibrosarcoma, and rarer tumors such as malignant schwannoma and malignant fibrous histiocytoma [2]. Most patients are asymptomatic, presenting with an indolent slow-growing mass. Unfortu-nately,theUSfeaturesofmanysolidextra-testicularmasses areoftenunspecific,precludingaspecificdiagnosisinmost cases[2].

Asstatedabove,leiomyomaoriginatesfromsmooth mus-cleandcanthereforebefoundinvariousorgans.With spe-cificreferencetothemalegenitourinarysystem,3different typesofleiomyomacanbeidentifiedonthebasisoforigin site;(1)derivationoferectorpilimuscle(piloleiomyoma),(2) derivationofsmoothmusclesofbloodvessels (angioleiomy-oma),and(3)genital leiomyoma(egfromthesmooth mus-clesofthescrotum)[4,5].Themajorityofmalegenitourinary tractleiomyomaisfoundintherenalcapsule,butthistumor hasalsobeenreportedintheepididymis,spermaticcord,and tunicaalbuginea[2,6].

Leiomyoma isaslow-growingtumor,frequentlyindolent withahigherincidenceofthefourthandfifthdecade,butthey canaffectallages.Usuallythepatientperformsanultrasound approximately7-8 years aftermasspresentation [7].AtUS

(3)

593

Fig.2– Coronal(A)andaxial(B-C)TurboSpinEchoT2-weightedimages;coronal(D)T1-weightedunenhancedandaxial(E-F) T1-weightedpostcontrastimages.MRimagesshowthepresenceinthescrotalsacofthe2heterogeneousparatesticular masses,1foreachside,noncapsulatedwithregularmargins.ThemasseswerehypontenseonT2-weightedimages, isointenseonT1-weightedunenhancedimages,andshowedheterogeneousenhancementaftercontrastagentinjection.

Fig.3– Grossspecimenofthelesionremovedfromleft scrotum.

examination,theyappearas solidhypoechoic or heteroge-neousmassesthatmayormaynotcontainshadowing calcifi-cation;thus,intheabsenceofcalcification,thistumormaybe indistinguishablefromadenomatoidtumorandfibroma[8].

MRI,however,accordingtoastudybyCassidyetal,isthemore sensitive and accurate imaging modality in detection and localizationofleiomyoma[9].Indeed,MRIallowstissue char-acterization,withitssignalintensitypropertiesallowing de-tectionoffat,bloodproducts,granulomatoustissue,and fibro-sis.ThetypicalMRIfeaturesofleiomyomaarethefollowing: (1)isointense signalonT1-weightedimaging,(2)lowsignal onT2-weightedimaging,and(3)lowercontrastenhancement comparedtoadjacentorgans,inthiscasethetestes[9].

Tothebestofourknowledge,thisisthefirstcasereport presentingimagingfeaturesofbothUSandMRIofbilateral paratesticularleiomyoma.Somepreviouscasesof paratestic-ularleiomyomahavebeenreportedintheliteratureandtheir characteristics are resumed in Table 1[10–13]. As emerges fromthereportedcases,thepreviouslydescribedleiomyoma haddimensionsrangingfrom1.1cmto5cmwithageatthe timeofthefindingbetweentheIVandtheVIdecadeoflife, generallyslowglowingandasymptomatic.OnUS examina-tiontheyappearedasmassesfrompredominantly heteroge-neousorhypoechoicechostructure,in1casetheypresented contextualcysticareas,thusinaccordancewithourfindings, themassshowedanhighlyheterogeneoustissue,the charac-teristicsofUSimagingareoftenunspecificanddonotallow adiagnosisofcertainty.Atpresent,surgicallocalresectionis stillthemainmethodforthetreatmentofleiomyoma;radical orchiectomyisnecessaryonlywhenthetumorisnot dissocia-blefromthetesticleorinpresenceofsignssuggesting malig-nantbehavior.Inconclusion,paratesticularleiomyomaisan extremelyuncommontumor,inmostcasesindolent, show-ingaslowgrowthandnoninvasivepattern.Timelydiagnosis

(4)

594

Table1– Casereportsof4patientswithbilateralparatesticularleiomyoma.Ageatdiagnosis,sizeoftumors,andUS appear-ancearereported;inthecasedescribedbyBremmeretal,1ofthelesionwasnotremovedduetothesmalldimensions.

Reference Ageatdiagnosis Sizeoftumors(cm) US

Fernandezetal(2017)[10] 56 3× 33× 3 Heterogeneous

Wangetal(2015)[11] 47 2.6× 2.21.4× 1.1 Hypoechoicheterogeneoustumor Bremmeretal(2012)[12] 59 5× 3.5 Echogenicandcysticareas Lia-Bengetal(1996)[13] 62 3× 31.5× 1.3 Lowechogenicity

andappropriate treatmentwouldlargelyprecludetheneed fororchiectomyinmostofthepatients.USisdefinitelythe modalityofchoiceforinitialevaluationofscrotalpathologic conditionsbecauseofitswideavailability,lowcost,andhigh sensitivityfordetectionofparatesticularmasses,butMRIcan helpimprovetissuecharacterization.

R E F E R E N C E S

[1] SiristatidisC, VaidakisD, RigosI, ChreliasG, PapantoniouN. Leiomyomaandinfertility.MinervaGinecol

2016;68(3):283–96.

[2] AkbarSA, SayyedTA, JafriSZ, HastehF, NeillJS. Multimodalityimagingofparatesticularneoplasmsand theirraremimics.Radiographics2003;23(6):1461–76.

[3] WoodwardPJ, SchwabCM, SesterhennIA.Fromthearchives oftheAFIP:extratesticularscrotalmasses:

radiologic-pathologiccorrelation.Radiographics 2003;23(1):215–40.

[4] NewmanPL, FletcherCD.Smoothmuscletumoursofthe externalgenitalia:clinicopathologicalanalysisofaseries. Histopathology1991;18(6):523–9.

[5] AganovicL, CassidyF.Imagingofthescrotum.RadiolClin NorthAm2012;50(6):1145–65.

[6]MakCW, TzengWS, ChouCK, ChenCY, ChangJM, TzengCC. Leiomyomaarisingfromthetunicaalbugineaofthetestis: sonographicfindings.JClinUltrasound2004;32:309–11.

[7]AlukoT, MasiZ, TomaszewskiJ, GermaineP.Scrotalsac leiomyoma:casereportofararebenignscrotalmass.Radiol CaseRep2018;13(2):411–14.

[8]HricakH, FillyRA.Sonographyofthescrotum.InvestRadiol 1983;18:112.

[9]CassidyFH, IshiokaKM, McMahonCJ, ChuP, SakamotoK, LeeKS, AganovicL.MRimagingofscrotaltumorsand pseudotumors.Radiographics2010;30(3):665–83.

[10]FernandezA, KrishnamoorthyS, MuralitharanS, JohnsonT, RamananV.BilateralSynchronousparatesticular

leiomyoma—arareentity.JClinDiagnRes.2017;11(4):PD05–6.

[11]WangAX, FengSL, ChangJW.Leiomyomaofthebilateral tunicaalbugineaoftestes:acasereport.IntJClinExpPathol 2015;8(8):9703–5.

[12]BremmerF, KesselFJ, BehnesCL, TrojanL, HeinrichE. Leiomyomaofthetunicaalbuginea,acasereportofarare tumourofthetestisandreviewoftheliterature.Diagn Pathol2012;7:140.

[13]Lia-BengT, Wei-WuangH, Biing-RornC, Chia-ChunT. Bilateralsynchronousleiomyomaofthetesticulartunica albuginea.Acasereportandreviewoftheliterature.IntUrol Nephrol.1996;28(4):549–52.