Van Trung Hoang
a,∗, Cong Thao Trinh
b, Trong Binh Le
b, Trong Khoan Le
baRadiologyDepartment,ThienHanhHospital,17NguyenChiThanh,BuonMaThuot,Vietnam
bRadiologyDepartment,HueUniversityofMedicineandPharmacy,Hue,Vietnam
a r t i c l e i n f o
Articlehistory:
Received22October2018 Revised4March2019 Accepted4March2019
Keywords:
Secondaryretroperitonealteratoma Maturecysticteratoma
a b s t r a c t
Maturecysticteratomaisoneofthemostcommontumorsoftheovaries,testis,medi- astinum,andretroperitoneum;however,secondaryretroperitoneumlesionsarerareen- titiesinadults.Wereportacaseofa 22-year-oldfemalewhowaspreviouslydiagnosed withamatureteratomaofleftovarianwashospitalizedduetodullabdominalpaininright hypochondria.Radiologicalevaluationrevealedamassintherightupperabdominaland flankregionwithanextensionfromtheposterioraspectoftheduodenum,composedof greasyandcysticelements.AtumorwasresectedthroughtheKocher’slaparotomyandthe pathologyreportconfirmedthediagnosisofamaturecysticteratomawithnoevidenceof malignancyorimmaturecomponents.
© 2019TheAuthors.PublishedbyElsevierInc.onbehalfofUniversityofWashington.
ThisisanopenaccessarticleundertheCCBY-NC-NDlicense.
(http://creativecommons.org/licenses/by-nc-nd/4.0/)
Introduction
Teratomasareuncommonneoplasmscomprisedofamixture ofdermalcellsderivedfromthe3germcelllayers(ectoderm, mesoderm, or endoderm), matureteratomas are rare neo- plasmscharacterizedbyinclusionofanywell-differentiated parenchymaltissues[1,2].Thesetumorsaremostcommonly foundintestesandovariesbutextragonadalsiteshavealso been reported including intracranial, cervical, mediastinal, retroperitoneal,andsacrococcygeal[3–5].Accountingforonly 4%ofallteratomas,retroperitoneallesionsarerareandmore commonamongchildrenratherthanadults[2,6].Wereporta
Funding:Thereisnotanyfundinourstudy.Althoughtypicalimagingfindingsofmaturecysticteratomasarewellknowntoradiologists, variousatypicalimagingfeaturescanbeparticularlymisleading.
CompetingInterests:Allauthorsdeclarethattheyhavenoconflictofinterest.
∗Correspondingauthor.
E-mailaddress:[email protected](V.T.Hoang).
caseofalargesymptomaticsecondaryretroperitonealmature cysticteratomaina22-year-oldwoman.
Case report
A22-year-oldwomanpresentedwithrighthypochondriapain.
Fouryearsago,sheunderwentsurgerytoremovetheleftovary dermatomecysts.Shebegantofeeluncomfortableandaching inthelowerrightquadrantforabout3monthsaftershefirst palpatedthelesion2and3monthsago.Recently,shenoticed anincreaseinthesizeofthemassandreferredtotheclinic.
Shedeniedanyassociatedsymptomsincludingfever,lossof
https://doi.org/10.1016/j.radcr.2019.03.008
1930-0433/© 2019TheAuthors.PublishedbyElsevierInc.onbehalfofUniversityofWashington.Thisisanopenaccessarticleunderthe CCBY-NC-NDlicense.(http://creativecommons.org/licenses/by-nc-nd/4.0/)
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Fig.1– Maturecysticteratoma(dermoid)ina22-year-oldwomaninwhomsymptomaticrighthypochondriamasswas palpatedataroutinephysicalexamination.Transversetransabdominalultrasonographyimageshowedacysticstructure (calipers)of112×110×65mmdimensionsinthemiddleoftheliverandrightkidney.Itwasheterogeneous,well circumscribedwithirregularborders.
appetite,weightloss,nausea,andvomiting.Shedeniedtak- inganymedications,smokingoralcoholconsumptionandher familyhistorywasunremarkable.
Herweightis53kg,gravidarum0,parity0.Shewasnor- motensivewithabloodpressureof110/60mmHg,herpulse ratewas72perminute,herrespiratoryratewas18perminute andhertemperature was37.5°C.Herphysicalexamination wasunremarkableexceptforafirm,nonmobilefullnesspal- patedinherrightupperquadrantregionwithoutanytender- nessorabdominalguarding.Theextentofthemasscouldnot beestablished.
Laboratory results showed that Ht, Hb, PLT, WBC, Glu, Protein,Urea, Cr,Na,K,and bHCG have fluctuated innor- malranges.Tumor markersAFP, CA15-3,CA19-9, CA125, andCarcinoembryonicantigen(CEA)havefluctuatedinnor- mal ranges. Serum levels of HBsAg were significantly ele- vated reaching the plateau of 5122ng/mL (normal ranges:
<0.9 ng/mL).Except that several urine parameters include urine-specificgravity(1.015),pH(9),protein(25mg/dL),leuko- cytes(500LEU/uL),anderythrocytes(25Ery/uL)weremildly changed.Ultrasonographyshowed a massof 112×110×65 mmdimensionsinthemiddleoftheliverandrightkidney.
Itwasheterogeneous,wellcircumscribedwithirregularbor- ders.Themassdid notappeartoarisefrom the rightkid- ney.Themasswasadherenttothesegment VIofthe right lobeof the liver. The mass has been described as a mul- tilocularmixture lesion.Doppler interrogated images were alsoobtainedandrevealedminimalinternalvascularity(Fig.
1).
A computerized tomography (CT) scan of the abdomen andpelviswithcontrastforthepatientthatrevealedalarge retroperitonealmasslyinginthemiddleoftheliverandright kidneymeasuringabout120×80×65mmindiameter.Itcon- sistedofboth cystic and solid elements,showing fat den- sitybutnotseencalcificationdensity.Noabnormalenhance- mentwasidentified. Themassdidnotinvadesurrounding organs(suchastheliver,therightkidney,therightadrenal
glands).Therewasnoretroperitoneallymphadenopathy,as- cites.The mass fixed to the broadclip ofthe old surgery (Fig.2).
Magneticresonanceimagingofupperandlowerabdomen hasalso beenperformed. Similarlytoultrasonography and CTscanreport,italsoshowslargemultilocularcysticspaces masswithmildenhancingasmoothwallandthinseptations as well asa little nonenhancing solid contents.The cystic hasbeenhemorrhagicheterogeneoussignalintensityinlo- cales withmany different phases.Note theabsence ofde- tectable calcificationormatted tuftofhairinmass(Figs.3 and4).
Laparotomywithexcisionoftheretroperitonealmasswas performedbytherightsubcostalline(Kocher’slaparotomy).A hugeretroperitonealmasswasobserved.Thesurgeonfounda tumorsectionsticktothesurfaceoftheliverandthestemof massisderivedfromtheretroperitoneal.Subsequently,com- pleteremovalofthemassfromtheliversurfacewassuccess- ful,andadhesionswerereleasedandthetumorwasseparated from thesurrounding tissue.Noasciteswere observedand basedonthefindingsadecisionwasmadefortotalexcision ofatumor.Finally,a12×8×6.5cmmasswastotallyexcised andsenttothelabforpathologicevaluations.Themasshad well-circumscribedsmoothbordersandrubberyconsistency.
Cut-sectionofthemassrevealedmultilocularcysticspaces, whitish-greywalls,scatteredyellowishadiposetissuecollec- tions,andmucussecretions.Thecystwallwasupto4-mm thick(Fig.5).
Histopathologic examination shows that within a neo- plasmcomposedofvarioustissuesincludingepithelialtissue, fat,cartilagetissuemixedwithfibroustissue,andmuscletis- suealongwithvastareasindicativeofpreviousbleedingwith aggregationofhemosiderin.Thefinaldiagnosiswasmadeas amaturecysticteratomawithnoevidenceofmalignancyor immaturecomponents(Fig.6).
Thepatientwasfollowed2weeksafterthesurgery,during whichshedevelopednosignificantcomplications.
Fig.2– CTscanthroughtheabdominalshowsamaturecysticteratomaintherighthypochondria.(A)Onthenon–enhanced CTaxialplaneshowsitcontainsfat(arrow).Presencenexttoatumorhasaclipoftheoldsurgerylostseats(arrowhead).(B) TheenhancedaxialCTinarterialphaseshowsboundofthemassisnotclearwithaliver(arrows).Thelesionshowswith mildhypervascular(arrowhead).(C,D)Thetumoronthesagittalandcoronalimagesinlateportalphase.
Fig.3– MRIofatumorintheaxialplaneonT2TSEsequence(A),T2TSEFS(B),non–enhancedT1VibeDixonsequence(C), enhancedT1VibeDixoninlatearterialphase(D),Diffusionweightedimaging(DWI)(E),andApparentdiffusioncoefficient (ADC)(F).Themassshowsamultilocularlesion(arrowsin(A)).Apartofthelesion(arrowhead)increasedsignalonT2W, increasedslightlyonT1W,andincreasedsignalonDWIandADC(T2shine-througheffect).Italsoshowsthesignal intensityofhemorrhageorfat(thinarrowsin(B)and(C)).
Discussion
Teratomasareacommonformofgermcelltumor,contain- ingall3germcelllayers.Teratomasareclassifiedasmature orimmature,dependingonthedegreeofdifferentiationofits components[7].Retroperitonealteratomasareuncommonin theadultpopulation.Primaryretroperitonealteratomasrep- resent1%-11%oftheretroperitonealneoplasms[1],butsec- ondary retroperitoneal teratomasare rarer and mostadult casesareinfemales.Thereareusuallyasymptomaticbutcan causenonspecificabdominaldisordersincludingpain,nau- sea,andvomiting.Inthemajorityofcases,theypresentas
asymptomatic,makingthediagnosisatanearlierstagemore difficult[8].Thisdisorderisimportanttodiagnosebecause 26%aremalignantwhentherearedetectedinadults[9].Ter- atomasinthislocationtendtobewelldeveloped.
Theretroperitonealteratomawasidentifiedbychancein thispatientandwascharacterizedbybenigncysticfeatures withgrease.Atransperitoneallaparoscopicresectionofatu- morwasperformed.Duringthesurgery,thetumorexhibited thesamephysicalappearanceandwasproximaltotheliver, andaswehadnotpreviouslyencounteredthissituation,com- plete removalofthe mass from the liver surfacewas per- formed.
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Fig.4– MRIofatumorinthecoronalplaneonT2Haste(A),T1-weightedin-phase(B),andout-of-phase(C)showcystic (asterisks)andgreasy(arrowheads)element.Onthenon–enhanced(D)andenhancedaxialT1-weightedimagesinthe arterialphase(E)andindelayphase(F)showthemixturesignalintensity(arrowheadsandthickarrows).Wallandseptals oftumorshowenhance(thinarrows).
Fig.5– (A)Photographofthegrossspecimenshowsthemasswithamultilocularlesion.Thestemofmassisderivedfrom theretroperitoneal.(B)Itshowsmultiseptatecysticmassfilledwithsebum,yellowishpastymaterialandseveralsolid nodularmassesfindingsthataccountforthefatechogenicityandsignalintensityseenatradiographyimaging.(Color versionoffigureisavailableonline.)
Inour case,a retroperitoneal mixedmasswas detected withultrasound.Thislesionwasinitiallyconsideredasapri- marytumor.Detailedanamnesisrevealedapreviousmature ovarianteratoma4yearspreviously,convertingthe initially primaryretroperitoneal massasahigh suspicion ofasec- ondarylocationoftheinitialteratoma,whichwasfinallycon- firmedbysurgery.Asinourcase,thereviewoftheliterature concludesthattheclinicalpresentationofsucharetroperi- tonealmassisnonspecificwithadullpainintherightupper abdominal.Theroleofimagingistoorientatethediagnosis.
Thepresentstudyreportedararecaseofaretroperitoneal teratomaaccompaniedbyabdominalpain,whichwastreated successfullybyacompleteresectionofatumor.Thepotential mechanismwasunclear,butwehypothesizedthatthe for- mersurgeryandsurgicalclipslostmayhavebeeninvolved.
Thediagnosisofaretroperitonealteratomacouldbemadeon thebasisofimagingstudies,andthegoldstandardtreatment strategyforthisneoplasmissurgicalresectionwithoutrup- ture.
Radiologycangiveinformationaboutthestructureofthe mass,whichcanbeof2types:solidorcystic.Ultrasound(US), CT,andmagneticresonanceimagingcontributetodetecting
the3componentsofthesolidtype(tissular,fatty,andcalci- fiedcomponents)andtoidentifyingthecystictype(withor withoutfatorcalciumcomponent).Thecystictypemaycon- tainenhancedseptations[9].Inourcase,thereistheabsence ofcalcification inmass.However,all other featuresare di- rectedtowardtheteratomawithoriginatingintheretroperi- tonealcavity.Thepresenceofthedifferentcomponentshelp tosuggestthecorrectdiagnosisonthebasisofimaging,but surgeryandpathologyarerequiredforthefinaldiagnosis.Dif- ferentialdiagnosisincludesasecondarylocation ofovarian carcinoma,sarcoma(whenfatispresent,thenliposarcoma hastobeevocated),ovarymetastases,neurogenicmass,tu- berculosis,Kaposisarcoma,Castlemandisease,andgrowing teratomasyndrome(developmentofamatureteratomafol- lowingchemotherapyforanonseminomatoustumor).
Whenweconsiderretroperitonealcysticmasses,thefol- lowingpossibilitiesaretobeincluded:theneoplasticcondi- tionsincludecysticlymphangioma,mucinouscystadenoma, cysticteratoma,cysticmesothelioma,Mülleriancyst,epider- moid cyst,tailgut cyst,bronchogenic cyst, pseudomyxoma retroperitoneum, and perianal mucinous carcinoma; non- neoplasticdisordersincludepancreaticpseudocyst,lympho-
Fig.6– Mixedgermcelltumors(maturecysticteratoma)ina22-year-oldwoman.(AandB,×10and×40,
hematoxylin-eosinstain)Photomicrographofthecystwallshowssquamouscelllining(arrowheads),sebaceousglands (arrows),andinterveningmuscle(asterisks).Sebaceousmaterialandcelldebrisareseenfillingthecystlumen(L)(×10and
×40,hematoxylin-eosinstain).(C,×40,hematoxylin-eosinstain)Theimageshowsthestructureofthemuscletissue.(D,
×10,hematoxylin-eosinstain)Photomicrographofcartilagestructure.
cele,,andhematoma[9,10].Athistology,fibrosis,necrosis,or matureteratomacanbeobserved[11].Inmostofthecases, surgicalismadeimmediately,withagoodoutcome.
Conclusion
Thesecondarylocalizationofretroperitonealteratomaisun- common.Althoughthediagnosiscanbemadepreoperatively bythecharacteristicsofatumorontheimagingmodalities, adefinitivediagnosis isestablishedupon histologicassess- ment.Imagingmodalitiescontributeforthesuggestionofthe correctdiagnosis,byshowingcystic retroperitonealmasses withseptation,multiplecalcifications,andparietalenhance- ment.Inpatientswiththepreviousstoryofovaryneoplasm, thesecondarylocationofthediseaseisoneofthediagnostic possibilities.Surgicalresectionisthemainstayinthetreat- mentofmatureretroperitonealteratomas.
Supplementary materials
Supplementarymaterialassociatedwiththis articlecanbe found,intheonlineversion,atdoi:10.1016/j.radcr.2019.03.008.
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