Successful adult-to-adult living donor liver transplantation using liver allograft after the resection of hemangioma: A suggestive case for a further expansion of living donor pool

CASE

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InternationalJournalofSurgeryCaseReports16(2015)166–170

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

Successful

adult-to-adult

living

donor

liver

transplantation

using

liver

allograft

after

the

resection

of

hemangioma:

A

suggestive

case

for

a

further

expansion

of

living

donor

pool

Yasuharu

Onishi

,

Hideya

Kamei,

Hisashi

Imai,

Nobuhiko

Kurata,

Tomohide

Hori,

Yasuhiro

Ogura

DepartmentofTransplantationSurgery,NagoyaUniversityHospital,Nagoya,Japan

a

r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received13July2015 Receivedinrevisedform 17September2015 Accepted30September2015 Availableonline8October2015

Keywords: Hemangioma Livergraft Adult-to-adult Livingdonor Livertransplantation Donorpool

a

b

s

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r

a

c

t

INTRODUCTION:Hepatichemangiomaisoneofthemostcommonbenignlivertumors.Therearefew publishedreportsregardinglivertransplantationusingliverallograftswithhemangioma.

PRESENTATIONOFCASE:A45-year-oldmanwasevaluatedasalivingdonorfor19-year-oldsonwith cirrhosisduetohepaticfibrosis.Preoperativeinvestigationsrevealed20and7mmhemangiomas,at seg-ment2(S2)and4(S4)respectively.ConsideringtheanatomicalrelationofS2hemangiomaandGlisson 2,livergraftwasdesignedasleftlobeexcludedS2hemangiomabypartialresection.Estimatedgraft recipientweightratio(GRWR)evenafterpartialresectionofhemangiomawasreasonable.Duringthe donoroperation,apartialhepaticresectionofS2hemangiomawasperformed.Intraoperative patho-logicfindingsrevealedacavernoushemangioma,andthen,thelefthepaticgraftwiththecaudatelobe washarvested.ActualGRWRwas0.90%.Donor’spostoperativecoursewasuneventful.Recipient’s post-operativecoursewasalmostuneventful.Postoperativecomputedtomographyoftherecipientshowed thegraftregenerationwithoutincreaseorrecurrenceofhemangioma.

DISCUSSION:Organshortageisamajorconcerninthefieldoflivertransplantation.Anoveldonorsource withafurtheroptionisextremelycrucialforaguaranteeoflivertransplantation.Weexperiencedthe firstcaseofadult-to-adultlivingdonorlivertransplantationusingliverallograftaftertheresectionof hemangioma.

CONCLUSION:Weadvocatethattheuseofliverallograftwithhemangiomasinadult-to-adultLDLT set-tingscanberemarkablestrategytoreducetheproblemoforganshortagewithoutanyunfavorable consequencesinbothlivingdonorandrecipient.

©2015TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction

Becausethe usageof extended criteria for donation of liver

transplantationhasbeenneededduetodonororganscarcityfor

transplantation [1],liver graftsfrommarginal donorsincluding

liverwithbenigntumorshavebeenacceptedastreatmentoptions.

Hepatichemangiomaisoneofthemostcommonbenigntumorsof

theliver,asdescribedupto7%inautopsyfindings[2].

Thehemangiomausually remainsasymptomatic[3] andhas

abenigncourse[4–6],althoughsymptomatichemangiomasmay

rarelyrequireeither interventional orsurgicaltreatment[7–9].

Abbreviations: LDLT,livingdonorlivertransplantation;GRWR,graftrecipient

weightratio;POD,post-operativeday;GV/SLV,graftlivervolumetostandardliver volume.

∗Correspondingauthorat:65Tsurumai-cho,Showa-ku,Nagoya466-8550,Japan. Fax:+81527442293.

E-mailaddress:onishiy@med.nagoya-u.ac.jp(Y.Onishi).

Therearefew publishedreports regarding livertransplantation

using liver allografts with hemangiomas [10–16]. In not only

deceased donorlivertransplantation butalsoliving donorliver

transplantation (LDLT), liver allograftswith hemangiomashave

beenutilizedfortransplantation.However,inLDLTsetting,there

areonlytwopublishedreportsaboutliverallograftswith

heman-gioma,andtheywerebothlivertransplantsforpediatricrecipients

[12,15].Untilnow,therehasbeennoreportinadult-to-adultLDLT

withlivergraftswithhemangioma.

We presentherethe firstreportof successfuladult-to-adult

LDLTusingliverallograftwithhemangioma.

2. Presentationofcase

A 19-year-old male was admitted with liver failure due to

congenitalhepatic fibrosis.Aprior diagnosisofcongenital

hep-aticfibrosis had been establishedby gastroenterologists before

13years. Hisliverfunctiondeterioratedprogressively,withthe

http://dx.doi.org/10.1016/j.ijscr.2015.09.043

2210-2612/©2015TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://

Fig.1. Preoperativecontrast-enhancedabdominalcomputedtomography(A,B)andmagneticresonanceimaging(C,D)ofthedonorliverwithcavernoushemangiomas.S2 hemangiomawasindicatedbyarrowheads,andS4hemangiomawasindicatedbyarrows.

following laboratory findings: serum total bilirubin 25.2mg/dl, prothrombintime-internationalnormalizedratio1.78,aspartate aminotransaminase119IU/l,andalanineaminotransferase71IU/l. Noruptureofesophagealandgastricvariceswasseen.However,he hadsomecriticalepisodesofgrade2hepaticencephalopathy,and intensivecaresincludingplasmaexchangewasseriouslyrequired. ThepreoperativeliverprofilewasevaluatedasChild-Pugh classi-ficationofgradeC(11points)andanMELD(ModelforEndStage LiverDisease)scoreof34points.

A45-year-oldmalewasevaluatedasalivingdonorforhisson. Theliverfunctiontestsofthedonorweretotallywithinthenormal rangewithnoevidenceofanycoagulopathy.Donorpreoperative computedtomographyandmagneticresonanceimagingrevealed 20mmand7mmhemangiomas,atsegment2(S2)and4 respec-tively(Fig.1).Estimatedleftlobevolumewiththecaudatewas

503ml.ConsideringtheanatomicalrelationshipofS2hemangioma

andGlisson2,livergraftwasdesignedastheleftlobegraftwith

caudatelobeexcludedS2hemangiomabypartialresection(Fig.2).

Estimatedgraftvolumeafterhemangiomapartialresectionwas

482ml,whichaccountedfor0.83%ofgraftrecipientweightratio

(GRWR).

Thus,therecipientstatuswasanend-stageliverdisease(i.e.,

advancedlivercirrhosis),andthisdonorwasonlyacandidatefor

thisrecipient.Afteranapprovalofinstitutionalethicalcommittee,

wescheduledLDLTforthiscase.

A LDLT donor and recipient procedure was performed as

describedelsewhere[17,18].Atthebeginningoflivingdonor

oper-ation,invivopartialhepaticresectionoftheS2hemangiomawas

performed without thePringle maneuver. Intraoperative

ultra-sonographywasusedtoidentifythelocationofS2hemangiomaand

Glisson2toavoidtheinjurytoGlisson2structures(Fig.3A).The

resectedspecimenfromthedonorliverwasconfirmedtobea

cav-ernoushemangiomabyanintraoperativepathologicexamination,

andthen,thelefthepaticgraftwithcaudatelobewasharvested.

Thedonoroperationtimewas521min,andthebleedingvolumeof

thedonoroperationwas1250ml,butmostofthebleedingoccurred

Fig.2. The3D-imagesimulation.HemangiomawasshowninH,withpartial resec-tionmargin.

aftertheresectionofthehemangioma.Theactuallivergraftweight

was504g,andresultinginanactualGRWRof0.90%.

Therecipientoperationwasperformedwithstandard

proce-dures.Atthetimeofreperfusion,nobleedingwasobservedfrom

theresectionsiteofS2hemangioma(Fig.3B).Splenectomywas

added in this case, because of the existence of splenic artery

aneurysm.Recipientoperationtimewas632min,andbloodloss

was5320ml.

Thedonor’spostoperativecoursewasalmostuneventful,andhe

wasdischargedfromthehospitalonthepost-operativeday(POD)

12.TherecipientwasdischargedfromthehospitalonPOD31

with-outsubsequentlivernecrosisorbileleakage fromtheresection

siteofS2hemangioma.However,onPOD39,therecipientwas

re-admittedtothehospitalbecauseoftheintra-abdominalbleeding,

andurgentoperationwasperformedforhemostasis.Thecauseof

bleedingwasgastricvarixruptureclosetosplenectomysite,and

heman-CASE

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Fig.3.(A)Donoroperation.Ultrasoundsonogramwasusedtoconfirmthelocation ofS2hemangiomaandGlisson2.Locationofhemangiomawasindicatedbywhite arrows.(B)Finalviewofrecipientoperation.NotenoGlisson2injuredafterpartial resectionofhemangioma.

gioma.Hewasdischargedfromhospital8weeksafterthesecond operationwithgoodgeneralconditionandlivergraftfunction.He iscurrentlydoingwell2yearsaftertheLDLT.Also,therecipient’sCT scansonthepost-operative132daysand19monthsshowedthat thegoodregenerationofthelivergraftwithoutanyrecurrenceor growthofhemangioma(Fig.4).

3. Discussion

Althoughlivertransplantationhasbeenwidelyperformedfor

end-stageliverfailure,organshortageisthegreatestproblem

fac-ingthefieldoforgantransplantationtoday.Therefore,theusage

ofextendedcriteriadonorsfororgantransplantationhasbecomea

necessityduetodonororganscarcityfortransplantation[1].

Actu-ally,theuseofmarginalorexpandedpooldonorswasshownto

haveoutcomesimilartoideallivergraft[19].Inrenal

transplan-tation,Khurram etal. reportedthatuseof kidneys aftertumor

resectionseemsafeasiblesourcetoincreasethedonorpool[20].

Thus,itisextremelyimportanttotrytouseanoveldonorsourceasa

furtheroptiontoincreasethenumberofpatientswhomightbeable

toreceivealivertransplantationaswellaskidneytransplantation.

Hepatic hemangioma is one of the most common hepatic

tumors.TheMayoClinicteamsuggestedthatmostliver

heman-giomacouldbeobservedsafely[4],andFargesetal.confirmedthe

statement[5].However,therearefewpublishedreportsregarding

livertransplantationusingliverallograftswithhemangiomasofar. Table

1 Review of the documented reports of liver allografts with hemangioma. Case no. Year Author Reference Deceased/living donor Donor age Recipient age Hemangioma Size Location Resection Size after transplantation 1 1995 Mor et al. [10] Deceased 54 30 10 × 10 × 8 cm Posterior segment Done at backtable Unknown 2 2005 Onaca et al. [11] Deceased 65 59 9 cm, 3 cm Right lobe, left lobe Done at backtable No recurrence 3 2005 Pacheco-Moreira et al. [12] Living 32 4 4.2 cm Lateral segment No Unknown 4 2006 Aucejo et al. [13] Deceased 65 53 10 × 8 × 9 Right lobe No Reduction 5 2007 Nikeghbalian et al. [14] Deceased 36 37 20% of whole liver Right lobe No Reduction 6 2011 Sanada et al. [15] Living 27 2 2 cm Lateral segment Done in operation No recurrence 7 2013 Sun et al. [16] Living (otherwise discarded) 41 27 7.8 cm Left lobe No No change Our case 2015 Onishi et al. Living 45 19 2 cm, 0.7 cm S2, S4 Done in operation, no No recurrence, no change

Fig.4.Post-operativeCTfindingsintherecipient.TheCTfindingsat132days(A) andat19months(B)wereshown.

AsshowninTable1,sevencasesofdonorliverswith

heman-giomas have been reported previously [10–16].There are only

tworeportsof leftlateralsegmentfromalivingdonor

contain-ing hemangioma transplanted to pediatric patients but not to

adultpatients[12,15]. WhileSunet al.indicated adult-to-adult

livertransplantationofanotherwisediscardedpartialliver

allo-graftwithacavernoushemangioma[16],therehasbeennoreport

aboutadult-to-adult LDLTusingliverallograftwitha cavernous

hemangioma.Toourknowledge,thisisthefirstcase

demonstrat-ingadult-to-adultlivertransplantationusinglefthepaticlobegraft

withcaudatelobeaftertheresectionofhemangiomafromaliving

donor.

Sanadaetal.haveproposeda strategyfortheusageofliver

allograftwithhemangiomasinpediatricLDLTsettings[15].They

proposedthattheresectionofhemangiomasdependsonthetumor

location,theestimatedgraftlivervolumetostandard liver

vol-ume(GV/SLV)ratioafterthetumorresection,andthetumorsize.

HemangiomasataperipherallocationandtheestimatedGV/SLV

ratioof40%andmoreafterthetumorresectionareconsideredto

beanindicationofsurgicalresection.Similartotheirproposal,we

resectedthe20mmhemangiomaofS2,becausethehemangioma

wasrelativelylargeratperipheralsite,andtheestimatedGRWR,

whichwe useinsteadofGV/SLVratio,aftertheresectionofS2

hemangiomawas0.83%.Ontheotherhand,smaller7mm

heman-giomaatS4wasnotresectedbecausethelocationwasrelatively

centralandthesizewassmaller.Thesizeandfeatureofthe

heman-giomainthetransplantedlivershouldbestrictlyfollowed,because

detailedlong-termedanalysisofliverallograftwithhemangioma

hasnotbeenperformedandapossibilityofsymptomaticchanges

mayremain.

Technicallyspeaking,inordertoresectthehemangioma,there

aretwosurgicalprocedures,backtableresection[10,11]orinvivo

resectioninthedonorsurgery[15].Wechoseinvivo resection

duringthedonoroperation,becausewebelievethatinvivo

pro-cedurecontainstheeasieranatomicalresectionofthetumorand

theshortercoldischemictimethanbacktableresection.

4. Conclusions

Our case is the first reportof adult-to-adult transplantation

usingalivingdonorlivergraftaftertheresectionofhemangioma.

Based onourexperience,donor liverwithhemangiomacan be

safelyusedevenfor adult-to-adultLDLT,whilethefollow-upis

neededincaseofthehemangiomaremainedinthetransplanted

liver.Weadvocatethatliverallograftswithhemangiomasshould

notbeconsideredasacontraindicationforadult-to-adultLDLT,and

canbeacceptedasapotentialliverallograft.

Conflictofinterest

Nofinancialconflictsofinterest.

Funding

Allauthorsdidnotreceiveanyfundingforthisreport.

Ethicalapproval

Thiscasereportiswrittenbasedoninstitutionalethical

com-mittee.

Consent

Writteninformedconsentwasobtainedfromthepatientfor

publicationofthisCasereportandanyaccompanyingimages.A

copyofthewrittenconsentisavailableforreviewbythe

Editor-in-Chiefofthisjournal.

Authorcontributions

YasuharuOnishicontributedreportsretrievalanddraftingof

this manuscript. Yasuharu Onishi, Hideya Kamei and Yasuhiro

Oguracontributedsurgicalproceduresofthiscasereport.Hisashi

ImaiandNobuhikoKuratacontributesdacquisitionofclinicaldata.

Tomohide Hori contributed critical revision of this manuscript.

Yasuhiro Ogura supervised this report. All authors read and

approvedthefinalmanuscript.

Guarantor

The guarantorof this manuscript is Yasuharu Onishi,

corre-spondingauthor.

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