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RenalLymphangiectasia,aRare
ComplicationAfterKidneyTransplantation
AghilèsHamroun1,2,PhilippePuech3,4,MehdiMaanaoui1,5,SébastienBouyé6,MarcHazzan1
andArnaudLionet1
1LilleUniversity,LilleUniversityHospitalCenter,DepartmentofNephrology,DialysisandKidneyTransplantation,Lille,France;
2NationalInstituteofHealthandMedicalResearch,CenterforresearchinEpidemiologyandPopulationHealth(CESP),Clinical
EpidemiologyTeam,Villejuif,France;3LilleUniversityHospitalCenter,DepartmentofRadiology,LilleUniversity,Lille,France;
4U1189-ONCO-THAI-ImageAssistedLaserTherapyforOncology,Lille,France;5INSERMU1190,TranslationalResearchfor
Diabetes,Lille,France;and6DepartmentofUrology,LilleUniversity,RegionalandUniversityHospitalCenterofLille,Lille,
France
Correspondence:AghilèsHamroun,LilleUniversity,LilleUniversityHospitalCenter,DepartmentofNephrology,Dialysisand
KidneyTransplantation,LilleF-59037,-mail:aghiles.******@
Received29December2020;revised4February2021;accepted1March2021;publishedonline30March2021
KidneyIntRep(2021)6,1475–1479;/
ª-
NC-NDlicense(-nc-nd//).
INTRODUCTIONCASE1
urgicalcomplicationsareanimportantissueafterA34-year-oldmanwasreferredtoourdepartmentfor
-
frequent,especiallylymphocele,lymphorrhea,orplantationforend-stagekidneydiseasesecondaryto
monthsaftertransplantationandaffectupto40%ofexperiencedabiopsy-provenacutesubclinicalhumoral
-
factors,
recognizedsuchasobesity,certainimmunosuppressivewastreatedwithcorticosteroids,intravenousimmuno-
drugs,,andimmunoadsorption(10sessions).The
beenimprovedalongwiththedevelopmentofcutting-,as
edgeimagingexaminationsandthewideuseofwellasurinaryalbumin-to-creatinineratioat50mg/
magneticresonanceimaging(MRI).3Upto15%ofwasthenmaintainedonacombinationoftacrolimus,
lymphocelesrequirespecifictreatment,themosteffi-mofetilmycophenolate,
cientoptionofwhichbeinglaparoscopicfenestration,appeared8yearsaftertransplantation,leadingtoan
giventhehighriskofrecurrenceaftersimplepercu-
Renallymphangiectasia,alsoknownasrenallym-,
–
phangiomatosis,57isanuncommoncomplicationafternon-chylous,andwithoutevidenceforinfectiousor
,
benign,ischaracterizedbyectasiaofperipelvic,peri-structuralabnormalityandpositronemissiontomography
renal,and/-scandidnotfindanyevidenceforcancerorlympho-
scriptionshavebeenreportedintheliterature,
exceptionallyafterkidneytransplantation,-
iscurrentlyonlysparsedataontheclinicalpresenta-formedanddidnotrevealanysignificantabnormality.
tion,pathophysiology,andtherapeuticoptionsinGiventhehypothesisofmycophenolate-inducedregen-
,whichmaynotbedetected
Wepresentheretwocasesofkidneytransplantonbiopsyfindings,aconversiontomammaliantargetof
lymphangiectasiarevealedbypersistentascitesandrapamycin(mTOR)inhibitor(mTORi)wasperformed.
atypicalabdominalpain,withoutkidneygraftMagneticresonanceurography(MR-urography)showed
(,603cc)withmultiple
KidneyInternationalReports(2021)6,1475–14791475
NEPHROLOGYROUNDSAHamrounetal.:RenalLymphangiectasia
-urographyfindingsregardingpatient1andpatient2.(a,b)Patient1.(c,d)
withmultipleplurilobularfluidcollectionsofdifferentmorphologicalpatterns:peripheraldevelopmentattheupperandlowerpolesofthegraft(patient
1:redarrows);parapyelic,juxtacapsular,andperihilar(patient2:yellowarrows).Themagneticresonanceurographiesalsoshowedlocalmechanical
complicationsofkidneygraftlymphangiectasia:largeinguinoscrotalhydrocele(redstar,b)andbladdercompression(yellowstars,candd).
lowerpolesofthekidneygraftaswellasextensiveascitesmTORiconversion,thepatientexperiencedbettercontrol
responsibleforinguinoscrotalhydrocele(Figures1aandofascitesandpainrelief,butthekidneygraftvolume
1b).Akidneygraftbiopsywasperformedandrevealedwasstillmoderatelyincreasing(,654cc).
chronicactivehumoralrejectionandsignificantintersti-Regardinggraftfunction,theserumcreatininelevel
tialedema,butnoargumentforpost--
,thetrans-geryundertheLichtensteinprocedurewasalsoproposed
plantedkidneyhadnormalappearanceatthetimeofforthetreatmentofinguinoscrotalhydrocele,butthe
,4yearspatientpreferredtodeclinetheproposition.
beforetherecentepisodeofascites(6yearsaftertrans-
plantation),anultrasoundscanwasperformedbecauseof
intermittentpaininthegraftarea:itssizehadalreadyCASE2
increased(13cm)andasignificantamountoffluid(4-mm-A35-year-oldmanwasreferredtoourdepartmentfor
thick)outlinedthegraft,whicharguedforperirenalchronicpelvicpainassociatedwithurinarysymptoms
lymphangiectasia(Figure2).Thisledusconcludeto8yearsafterfirstkidneytransplantationforend-stage
1476KidneyInternationalReports(2021)6,1475–1479
AHamrounetal.:RenalLymphangiectasiaNEPHROLOGYROUNDS
and1d)andmofetilmycophenolatewasswitchedto
,graft
sizesignificantlyincreased6monthslater(ÂÂ
18cm,910cc).mTORiwasthenwithdrawnandmofetil
,thekid-
neygraftvolumepartlydecreased(17cm,770cc)but
ascitesreappeared.
DISCUSSION
Wedescribetwocasesofkidneygraftlym-
(13phangiectasiaresponsibleforpersistentascitesand
cm)andathinperirenalhypoechoiclayer(),suggestiveofsymptomaticnephromegalyrevealedbyatypical
:
inguinoscrotalhydrocele(patient1)andbladder
kidneydiseasesecondarytotypicalchildhoodhemo-compression(patient2).
-Renallymphangiectasiaisanextremelyrarepa-
plantation,heexperiencedabiopsy-,almost
acutehumoralrejection(
becauseofdenovodonor-specificantigen).Therefore,-
heunderwentatreatmentcombiningcorticosteroids,neylymphaticvesselsandlargeretroperitoneallym-
intravenousimmunoglobulins,andsixplasmaex-
-
,contrib-
albumin-to-creatinineratioat32mg/-andperinephric
,lymphaticvesselsare
tacrolimus,mofetilmycophenolate,,unliketheme-
therecentsymptoms,,9Thedevelopmentoflymphaticvesselsis
performedandfoundasignificantnephromegaly(16controlledbypro-lymphangiogenicfactors(vascular
cm),associatedtonormalpyelocalicealcavities,multi-endothelialgrowthfactors[VEGF]CandD)bindingto
pleperipyeliccysts,perihepatic,andperisplenicasci-,only
,therewasnosignofonepreviousreporthasdescribedlymphangiectasiain
-fullyexplainthepathophysiologyofthedisease
formedanddidnotfindanyevidenceforperitonealbecauseanastomosisofthelymphaticvesselsisnever
lymphomatousdisease,,
-venacavavenographyanothercauseofmechanicallymphaticobstruction
,MR-shouldalwaysbeexcluded:inthecaseofpatient2,for
urographyandMR-angiographyofthekidneygraftexample,MR-angiographywasalsoperformedgiven
confirmedmajornephromegaly(ÂÂ17cm,-
729cc),multipleparapyeliccysts,pericapsularlym-dispositionhasbeensuggestedbuttheoutcomeofthe
phangiectasias,-
fat,thewholebeingresponsibleforbladdercompres-theless,themorphologyofthesekidneytransplants
,atthetimeoftransplantation,,the
,
peripyeliccysthadbeendescribedatthetimeoflymphangiogenesisduetopro-inflammatoryevents,
transplantation,noronanotherultrasoundperformed2suchasacuterejectionoracutekidneyinjury,appears
,thesesituationsprecisely
todiagnosesubclinicalacutehumoralrejection3yearspromotingVEGFC/VEGFDsecretionbycorticaland
aftertransplantation,,8,9Becauseofthe
basisofimagingoutcome(especiallyMR-urography),steadyprogressinthemanagementofkidneygraft
thediagnosisofkidneygraftlymphangiomatosiswithrejectioninthepastfewyears,thismayalsoexplain
aspecificperihilardevelopmentwasmade(Figures1cwhythispathologicalentityhasonlybeenrecently
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NEPHROLOGYROUNDSAHamrounetal.:RenalLymphangiectasia
Consideringatypicalascitesandnephromegaly,wetransplantation.
Thisexclusiondiagnosisshouldbesuspectedincaseofmultipleperirenaland/or
wereprimarilyconcernedaboutthediagnosisofpost-peripyeliccysts,responsibleforprogressivenephromegalyandatypicalascites.
patientshadbeentransplantedforalmost10yearsandapproach.
bothgraftswereprovidedbyyoungdonors(23and28Theprognosisismostlydrivenbylocalmechanicalcomplicationsand
alterationofqualityoflife.
yearsold,respectively).Interestingly,inthetwocasesThereisnospecifictreatmentandnephrectomymaybejustifiedincaseofpooroutcome.
previouslydescribedbyDawideketal,.7thedonors
,activelesionsmarsupializationwasalsoraisedbutnotretainedgiven
associatedtochronichumoralrejectionwerestillpresentitslowexpectedyield:indeed,unlikelargeclassical
,asmentionedabove,alymphocele,kidneylymphangiectasiaaremadeofa
chronicinflammationstatemighthavepromotedlym-complexnetworkofmultiplesmallcysticcollections
.
nativekidneys,whichisoftenassociatedtokidneyBecauseoftheunsuccessfulexperiencereportedby
dysfunction,,andthe
transplantpatientsdespit