Leiomyomatosis Peritonealis Disseminata (LPD) ten years after laparoscopic myomectomy associated with ascites and lymph nodes enlargement: a case report

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InternationalJournalofSurgeryCaseReports25(2016)1–3

Contents lists available atScienceDirect

International

Journal

of

Surgery

Case

Reports

j o u r n a l h o m e p a g e :w w w . c a s e r e p o r t s . c o m

Leiomyomatosis

Peritonealis

Disseminata

(LPD)

ten

years

after

laparoscopic

myomectomy

associated

with

ascites

and

lymph

nodes

enlargement:

a

case

report

Luigi

Nappi

_,

_Felice

_Sorrentino

_,

_Stefano

_Angioni

_,

_Alessandro

_Pontis

_,

_Ida

_Barone

_,

Pantaleo

Greco

a_{InstituteofObstetricsandGynaecology,DepartmentofMedicalandSurgicalSciences,UniversityofFoggia,Foggia,Italy} b_{InstituteofObstetricsandGynaecology,DepartmentofSurgicalSciences,UniversityofCagliari,Cagliari,Italy}

c_{DepartmentofMorphology,SurgeryandExperimentalMedicine,SectionofObstetricsandGynaecology,UniversityofFerrara,Ferrara,Italy}

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received28April2016

Receivedinrevisedform15May2016 Accepted17May2016

Availableonline2June2016

Keywords: Ascites

Leiomyomatosisperitonealisdisseminata Electricalmorcellation

Myomectomy

a

b

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INTRODUCTION:Wereportacaseofleiomyomatosisperitonealisdisseminata(LPD)arising10yearsafter alaparoscopicmyomectomythatwasassociatedwithascitesandlymphnodesenlargement.

PRESENTATIONOFCASE:Thepatientpresentedwithsmalluterinefibroidswithadominantposterior intra-muralfibroidmeasuring9cmindiameterandnormalDoppler.Laparotomyrevealedauterusenlarged withfibroids,whichwasdenselyadheringtotheurinarybladder,greateromentum,andsigmoidcolon. Multipletumorsofdifferentsizewerefoundattachedtotheperitoneum,omentumandbowel.The histopathologicexaminationwasconsistentwithleiomyomatosisperitonealisdisseminata.

DISCUSSION:Piecesofsmoothmusclecelllostintheabdominalcavityduringelectricalmorcellationafter laparoscopicmyomectomymayprogresstoleiomyomatosisperitonealisdisseminataevenaftermany years(teninourcase)anditcanbeassociatedwithascitesandlymphnodesenlargement.

CONCLUSION:Thisisthefirstcasereportedinliteratureofleiomyomatosisperitonealisdisseminatawith theseparticularfeatures(timeofclinicalpresentation,ascites,lymphnodesenlargement).

©2016TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction

Leiomyomatosisperitonealisdisseminata(LPD)isarare pathol-ogy of unkown etiology characterized by the appearance of multiplepelvicandabdominalnodules,whicharebasically com-posedofsmoothmuscle.Atpresent,around100casescanbefound inliterature.Wepresentthefirstcaseofapatientwhodeveloped disseminatedperitonealleiomyomatosistenyearsaftera laparo-scopicmyomectomythatwasassociatedwithascitesandlymph nodesenlargement.

2. Casereport

A40-year-oldmultiparouswomanwasadmittedtoourUnitin May2014withincreasingmenorrhagia,pelvicpressure,abdominal pain anda 2-years history ofpolycythemia. Laboratory evalua-tionrevealedanhemoglobinvalueof18.0g/dL,redbloodcellsof

∗ Correspondingauthorat:InstituteofObstetricsandGynaecology,Department ofMedicalandSurgicalSciences,UniversityofFoggia,VialePinto,71100Foggia, Italy.

E-mailaddress:felice.sorrentino@alice.it(F.Sorrentino).

6.15×106_{/␮Landerythropoietinlevelof18mU/mL.Thepatient’s}

historyincludedalaparoscopicmyomectomywithelectrical mor-cellationperformedin2004inourUnitandahysteroscopycarried out for heavy and prolonged menstrual loss 3 months earlier showednofocal lesionwithnoabnormality on histopathologi-calexaminationoftheendometrialbiopsyobtainedatthetime. Therewasnohistoryoforalcontraceptiveuse.Aftercheckingthe clinicalnoteswefoundthatthepatientunderwentlaparoscopic removalofasingle,anterior,peduncolatedmyomaof6cmthatwas morcellatedduringremoval.OnadmissioninMay2014,apelvic ultrasound scan showedmultiple small uterinefibroids witha dominantposteriorintramuralfibroidmeasuring9cmindiameter andnormalDoppler.Computedtomographyscansrevealed multi-plepelvictumorscloselyattachedtoperitoneumandbowelwith heterogenous enhancementofthemasses atcontrast-enhanced images.Additionally,ascitesandslightlyenlargedparaaorticand iliaclymphnodeswerefound.Standardtumormarkerswerewithin normallimits:Carcinoembryonicantigen(CEA)1,1ng/ml;Cancer Antigen125(CA-125)20,8U/ml;CancerAntigen 15-3(CA15-3) 22,5U/ml;carbohydrateantigen19-9(CA19-9)8,3U/ml; Alpha-fetoprotein(AFP)3IU/ml.Thesurgicalprocedure(typeofincision, risks,possiblecomplications...)wasexplainedbyourconsultant andinformedconsentfortotalabdominalhysterectomyand

bilat-http://dx.doi.org/10.1016/j.ijscr.2016.05.017

2210-2612/©2016TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://

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2 L.Nappietal./InternationalJournalofSurgeryCaseReports25(2016)1–3

Fig.1.Myomaattachedtothebowel.

eralsalpingooopherectomywassignedbythepatienttakinginto consideration she had completed her family. Laparotomy with midlineincisionrevealedauterusenlargedwithfibroidsstrictly adherenttotheurinarybladder,greateromentum,andsigmoid colon.Anasciticfluidsampleforexaminationwascollected. Multi-pletumorsofdifferentsizewerefoundattachedtotheperitoneum, omentumandbowel(sizebetween0,5and3cm)(Fig.1).The omen-talandthebladderadhesions werereleased.The noduleswere dissectedandremovedusingaLigasureAtlas.Parasiticmyomas wereremovedjustopeningawindowintheperitoneumand gen-tly pulling out the pathology by using a grasping forceps. The peritonealwindow was closed usingVicryl 2-0. On the bowel, whennecessary,stichesontheserosawereapplied.Hemostasis wasachieved using bipolaror stitches (Vicryl 2-0 or 3-0). The histopathologicexaminationwas consistentwith leiomyomato-sisperitonealisdisseminata.Therewasanycytologicalevidence ofmalignanceafterasciticfluidsampleexamination.No intraop-erativeorpostoperativecomplicationswerereportedandpatient wasdischarged3days aftersurgery. Twomonthsaftersurgery, thepatientwasasymptomaticwithahemoglobinof13.9g/dLand erythropoietinlevellessthan4.0mU/mL.

3. Discussion

Thecaseofa patientwhodeveloped LPDseveralyearsafter a laparoscopic myomectomy when electrical morcellation was carriedoutistimelybecauseoftheongoingconcernsabout morcel-lation.LPDisarareconditionfirstdescribedin1952,characterized bythepresenceofmultiplesmoothmusclenodulesthroughout theperitonealcavitywhichappearsgrosslymalignantbut histo-logicallybenign[1,2].Somerarecasesofmalignanttransformation havebeenreportedespeciallyinpostmenopausalwomen[3].The etiology isunknown, but it is thoughttooriginatefrom meta-plasiaofsubmesothelial, multi-potentialmesenchymalcells [4]. An associationwith high levels of exogenous and endogenous femalegonadal steroidshas beenfound[5]. However,in some

casesalinkbetweendisseminationofuterinetissueduring elec-tricalmorcellationand developmentof LPDhasbeen proposed

[6,7].Probablyduringelectricalmorcellationpiecesofspecimens aredispersedthroughouttheabdominalcavity;thentheybecome implanted into normal tissue and give rise to development of fibrotic nodules.Laparoscopicin-bagmorcellation offibroidsor uterushasthepotentialtopreventtheraremorcellation-related complications typicalof “open”morcellation inabdominal cav-ity[8–10].Patientsmaypresent mostlynon-specificsymptoms, suchasirregular,heavyuterinebleeding,painordiscomfort, gas-trointestinal bleeding and peritonitis (following erosionof LPD implantsinthebowelwall).Inourcasea2-yearhistoryof poly-cythemiawasalsodescribed.Theassociationbetweenlargeuterine myomasandsecondarypolycythemiahasbeenpreviouslyreported but themechanism is not clear (erythropoietinoverproduction is oneof thepostulatedtheories) [11].Finaldiagnosisis based onhistologicalexamination.Therelationshipwithmyoma mor-cellationremainsunclear.However,ourcasehighlightstheneed toaccuratelyremoveallmorcellatedfragmentsofmyomas dur-ingprocedureandthelengthoftime thatelapsedbetweenthe laparoscopicmyomectomyperformedin 2004and thepatient’s presentationwithdisseminatedleiomyomatosis:tothebestofour knowledgethisislongerthaninpreviousstudies.Also,theascites withlymphnodeenlargementwhichcanraisesuspicionof malig-nancyareworthemphasizing(theassociationofLPDwithascitesis veryrareanditspresencedoesnotexcludeLPDfromthe differen-tialswhenabdominalandperitonealmassesarefound).Obviously, alaparoscopicsurgerycouldalsohavebeenperformedbutwe pre-ferredtostartwithalaparotomyfortheparticularfeaturesofthe case.

Conflictsofinterest

Theauthorshavenoconflictsofinteresttodeclare.Thisstudy wasnotsupportedbyanypersonorinstitution.

Funding

Thisstudywasnotsupportedbyanypersonorinstitution.

Ethicalapproval

None.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereport.

Authorscontribution

LuigiNappi,FeliceSorrentino,IdaBarone:studyconceptand design. Felice Sorrentino, Stefano Angioni, Alessandro Pontis: acquisitionofdata.FeliceSorrentino,StefanoAngioni,LuigiNappi: draftingofthemanuscript.PantaleoGreco,StefanoAngioni:critical revisionofthemanuscriptforimportantintellectualcontent.

Guarantor

FeliceSorrentino

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