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2021Vol.7 No.1:4

Case Report

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Gynaecology & Obstetrics Case report ISSN 2471-8165

Wong LP1*, Jesani JK2, Namasaka WR1 and Wilson WK3

1 DepartmentofReproductiveHealth,CollegeofHealthSciences,MoiUniversitySchoolofMedicine,Eldoret,Kenya

2 DirectorateofReproductiveHealth,MoiTeachingandReferralHospital,Eldoret,Kenya

3 ChiefExecutiveOfficer,MoiTeachingandReferralHospital,Eldoret,Kenya

*Corresponding author: WongLP

unefemme30@gmail.com

Resident,DepartmentofReproductiveHealth,MoiUniversity,CollegeofHealthSciences,SchoolofMedicine,P.O.Box4606-30100,Eldoret,Kenya.

Tel: 254712913962

Citation: WongLP,JesaniJK,NamasakaWR, WilsonWK(2021)PostpartumRuptureofRightInternalIliacArteryAneurysmCaseReportfromaTertiaryHospitalinKenya.GynecolObstetCaseRepVol.7No.1:4

IntroductionIliac artery aneurysm is an extremely rare vascular disorderoccurring in about 0.03%of the population [1]. Approximately30% to 50% are bilateral [2,3]. The iliac arteries are normallysituateddeep in thepelvisand, itdivides into the internalandexternaliliacarteries,hencethediagnosisofaneurysmsofthesevesselsaredifficult.Theyaredetectedwhentheyareenlargedorrupturedorasanincidentalfindingduringimagingstudiesforotherabdominalpelvicdisorders.

Spontaneousruptureoftheinternaliliacarteryinpregnancyorpostpartumisrareandhasalethaloutcome,itwasfirstdescribedin1778[4].Tillcurrentdate,theexactetiologyofitisunclearanditisbelievedtobemultifactorial.

Some hypothesize that disorders such as endometriosis,congenital diseasesof connectivetissue (suchas Ehlers-Danlossyndrome type IV), and congenital malformations of bloodvesselssuchasaneurysm,includinganeurysmoftheinternaliliacartery,mayresultinarterialruptureinpregnancy,postpartumorinnon-pregnantpatients[5-10].

Incidences of aneurysm maybe found in 0.1% of general

population,withaneurysmoftheiliacarteryaccountingfor1%ofthesecases.Commoniliacarteryis involvedin85%,internaliliacarteryin10%andexternaliliacarteryin1%ofcases[11-13].

High blood pressure in the uterine or ovarian arteries duringmuscleactivityandstrainduringpregnancyordelivery isariskfactor for rupture of the artery [14,15]. Differential diagnosisthatmay contribute to ruptureof aneurysmmaybeprematureplacenta abruption, spontaneous uterus rupture, and spleenrupturei.e.,aneurysmofsplenicarteries.

Nevertheless,nonspecificsymptomsofarteryrupturemayresultin unrecognizable severity and urgency of the problem, whichleads to life threatening circumstances to themother and thechild[16].Themortalityofmotherswithspontaneousruptureofuterusarteryhasdramaticallydecreasedfrom49%to3.6%owing

Received: November16,2020; Accepted: February09,2021; Published: February17,2021

Postpartum Rupture of Right Internal Iliac Artery Aneurysm Case Report from a Tertiary

Hospital in Kenya

AbstractIliac artery aneurysm is an extremely rare vascular disorder occurring in about0.03%ofthepopulation.Suddenhemodynamicchangesalongwiththeradiologicalfindings of free abdominal fluid warrants for further surgical intervention, asthe heavily gravid uterus during pregnancy that compresses on the aneurysmundergoesasuddenreductionofsizepostpartumandtheassociatedchangesinpressuregradienton thearterypost-deliverymaycausesuddenruptureof theiliacarteryaneurysm.Ruptureoftheinternaliliacarteryisararecomplicationinpregnancyandinpostpartumbutshouldbeconsideredasadifferentialdiagnosisofabdominalpainpost-partum.Wearepresentinga29-year-oldpara3+0whodeliveredviaSVDwithnormalantenatal,intrapartumandimmediatepostpartumvitals.Onday1Post-Partumthepatientwentintocardiovasculararrestsecondarytohypovolemicshock,thepostmortemrevealedrupturedrightinternaliliacarteryaneurysm.

Keywords: Rupturedinternalrightiliacartery;Aneurysm;Post-mortem

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Gynaecology & Obstetrics Case report ISSN 2471-8165

hematomaontheboardligamentwithnormalpostpartumuterus(Figures 4 and 5).TheaortaandleftilliacarterywasnormalbutfoundarupturedRight internal iliacarteryaneurysm.Allotherinternalorganswerenormal.

to improvement of intensive intraoperative and postoperativetreatment[17].Earlydiagnosis,radiologicalstudiesandintensivesurgical-anesthetictreatmenthelpsinpreventionofmortality.

Wepresentacaseofincidentalpostmortemfindingofarupturedrightinternaliliacaneurysminapostpartumwoman.

Case ReportA 29-year-old Para 2 gravida 3 was admitted into a TertiaryHospital with complaints of lower abdominal pains for 1 dayat 43 weeks gestation by her LNMP, She perceived normalfetal movements. She had no history of any chronic medicalconditionsorpriorhistoryofsurgeries.Sheattendedantenatalclinics throughout her pregnancy, her antenatal profile wasunremarkable. She had history of postpartum Hemorrhagein her previous two pregnancies. On admission, the patientwas in FairGeneral Conditionwith vitalswithin normal ranges(BP - 133/82mmHg, Pulse - 106 beats perminute, Respiratoryrate - 20 perminute, Temperature - 35.7, SpO2 - 100%). Therewasalsonopallorongeneral examination.AntenatallyherHBwas 12.8 g/dl. Hemodynamically, the patient was stable. Onabdominalexamination-therewasnotendernessonpalpation,thefundalheightwasterm,fetus incephalicpresentationwitha longitudinal lie, fetal heart rate was 140 beats per minute.Onvaginal examination-The cervixwas5cmdilated, thick andcentrallypositioned.Theothersystemswereessentiallynormal.ShewasadmittedwithaDiagnosisofActivephaseoflabor.ThePatientprogressedwellaccordingtostrictpartographmonitoringandsuccessfullydeliveredaLiveMaleInfant,3.6kgwithagoodApgarscore.Post-partumvitalswerewithinnormalranges.Twohourspost-delivery,patientwastakentothenormalpostpartumwardfor12hoursmonitoring.Onthefollowingday,thepatientwas fit for discharge, as shewas leaving theward she startedcomplaining of dizziness and she collapsed, on examinationthe patientwas very pale, sweatywith cold extremities. Vitalswere86/60,pulse-146beatsperminute,randombloodsugar-7.0mmol/L.Abdominalexaminationsrevealedrightiliacfossatenderness,theuterinefunduswasnotwellappreciated.Vaginalexaminationwasnormal.An impressionofHypovolemic Shocksecondary toPrimaryPost-PartumHemorrhagewasmade.Shewas resuscitated with intravenous fluids, one unit of wholeblood, and a Pelvic ultrasound was done immediately whichrevealed a right iliac fossa hematoma (measuring 210 cm3)(Figure 1), with minimal ascites noted (hepato-renal space)(Figure 2) Full hemogram showed Hemoglobin of 12.8 g/dL,Plateletsof273×103/uL.CTscanoftheabdomenandpelvicwassuggestedforfurtherevaluationbutpatientwasunstabletobetakenforthescan.Decisiontoperformexplorationlaparotomywas made, however patient started gasping upon arrival tothe operation theater. Cardiopulmonary resuscitation as perNational ACLS protocol was done, however resuscitation wasunsuccessful.Clinicalpostmortemwasperformed,morethan2Lofhemoperitoneumfoundintheabdominalcavity.Largeamountof blood clot seen in the retroperitoneum space spreading allthe way to the diaphragm (Figure 3). There was a soft tissue

Ultrasoundfindingofrightiliacmass.Figure 1

Minimalascitesnotedinthehepato-renalspace.Figure 2

Largeamountofbloodclotsintheretroperitonealspace.Figure 3

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Gynaecology & Obstetrics Case report ISSN 2471-8165

Discussion In this patient, the exact etiology of the spontaneous arterialrupture was not established. We believe that there might bean existing arterial pathology, but we were not able to showevidences of any arterial pathology existence with eitherhistological or radiological examination. The pathogenesis ofiliac artery aneurysms is multifactorial and similar to that ofAbdominal Aorta Aneurysm (AAA). Etiologies that have beenreported to contribute to iliac aneurysmal dilatation includeatherosclerotic changes, inflammation, wall stress and walltension, proteolytic degradation of arterial wall tissue, andmoleculargenetics.Histologically,aniliacaneurysm,similartoanAAA,ischaracterizedbymacrophagedegradationofthemedialelastin lamellar architecture by metalloproteinases. The lesscommonetiologiesthatmaycauseInternaliliacArteryAneurysm(IIAA) include trauma, fibromuscular dysplasia, infection, cysticmedialnecrosis,collagenvasculardiseasesandpregnancy[18].

There is an ongoing debate as to whether pregnancy maycontributetothepathogenesisofvasculardisease.Cardiovascularchangesduringpregnancymayenhancethestressonthearterialwall.Highlevelsoffemalehormonesduringpregnancyalterthe

histological structureof thearterialwall, thereby resulting inapredisposition to aneurysmal dilatation. Pregnancymay be aninitiatorofarterialdegeneration,whichisadditivewithmultiplepregnanciesandchangesmaybepermanent[19].

Aneurysm rupture during pregnancy is often associated withnonspecific symptoms and is life-threatening. The maternalmortalityrateforvasculardissectionandruptureis0.74–0.76per100,000livebirths.Itisimportanttohavedifferentialdiagnosesin mind as this contributes to the complexity of diagnosingthis condition. Differential diagnoses include: uterine rupture,placentalabruption,massivepulmonaryembolism,cholecystitis,perforatedulcer,andurolithiasis[20].

Diagnosis canbemadeby computed tomographyangiography,MRA and abdominal/transvaginal ultrasound, which is quick,easyandfreeofionizingradiation.USisusedextensivelywithinobstetrics and gynecology and many clinical situations arediagnosedviaacombinationofclinicalexperienceandultrasoundskill.Adetailedevaluationofthekidneys(size,location,tumors,stasisandconcrements),aspecificdescriptionoffluidcollections(freefluid,hematoma, serous,mucinousetc.) anduseof colorflowmappingprovidedvaluableinformationforexactdiagnosisofanextremelyrareconditioninthiscase.

Iliac artery aneurysm has a potential to cause spontaneousruptureandcommonlypresentswithaneurysmoftheabdominalaorta,withtheincidenceof10%.Isolatedfindingofinternaliliacarteryaneurysmisrare,withtheincidenceof2%[21-23].Whilesingleiliacarteryaneurysmisuncommon,accountingforaround0.03%ofcases. It isexpressedasenlargementof theartery todoublethesizewithoutanysignsofanyotheraneurysminotherlocations[24-27].

Limited amount of literature concerning internal iliac arteryaneurysm or rupture in pregnant or postpartum women wasfound. However, pre-rupture finding of aneurysm can bemanagedwithelectivesurgery,thusreducingthemortalityrateto less than 10%. Endovascular techniques have become themainstayinIIAA'streatment.EndovasculartreatmentofIIAAcanbebasedonapureendovascularapproachorahybridoneaspartof an endovascular aneurysm repair (EVAR)procedure to treatanaortoiliac aneurysm [compromising internal iliac artery (IIA)patencybilaterally],inwhichabypassisperformed,usuallyfromthedistalexternaliliacarteryorthecommonfemoralartery,torevascularizeoneoftheinternaliliacarteries[28].

Pure endovascular techniques may be allocated in two mainphilosophies: with or without IIA flow preservation. Whilethe second option is inarguably easier to perform, it can beassociated with ischemic complications, especially in case ofbilateralIIAocclusion.Theworstofthesecomplicationsincludegluteal/perineal skin necrosis [29,30], colon ischemia [31], andspinal cord ischemia [32]. Despite representing amore benigncomplication, the incidence of buttock claudication can reachupto28%inpatientswithunilateralocclusionofIIAand42%inpatientswithbilateralIIAembolization[33].

Inordertoachieveinternaliliacarteriesflowpreservation,severalendovascular techniques were developed, including externaliliac to internal iliac artery stent grafts coupledwithaorto-uni-

Softtissuehematomaontherightboardligament.Figure 4

Normalpostpartumuterus.Figure 5

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Gynaecology & Obstetrics Case report ISSN 2471-8165

iliacrepairs,"sandwich"stentgrafts,andiliacbranchstentgrafts(IBSGs). An intra-peritoneal rupture are commonly associatedwithsuddendeathandhasextremelypoorprognosiscomparedtoRetroperitonealruptures.

Acuteabdominalpainscanbeasignofarterialrupturecausingacute bleeding along with hemodynamic changes. Symptomsbecomemoreprominentafterdeliveryastheextensivelygraviduterus undergoes sudden decrease in size and weight causingsudden arterial rupture due to the pressure changes in thevessels.Anysignofabdominalpain inpregnantorpostpartumwomanshouldbetakenseriouslyalongwithradiologicalfindingof free liquid in the abdominal cavity and immediate surgicalintervention is warranted. To increase awareness of this rarecondition,wereportthiscasewhichisanimportantdifferentialdiagnosistoconsiderinpregnantwomenwithabdominalpain.

ConclusionRupture of the internal iliac artery is a rare complication inpregnancyandpostpartum.Abdominalpainofunknownoriginwillbe importanthallmark indiagnosisofarterial rupture.Pre-rupture Iliac artery aneurysm diagnosed early in pregnancy

couldreducemortalityandmorbidityrisktoless10%.Bleedingbecomesmore severe intra-operatively when the compressivefactor of the gravid uterus is removed. Since rupture of theinternaliliacarteryisanextremelyrarecomplicationinpregnancyorduringpostpartum,neverthelessitshouldbeconsideredasadifferentialdiagnosisofabdominalpainorsuddenhemodynamicinstability.

Conflict of InterestTheauthorsdeclarethatthereisnoconflictofinterests.

AcknowledgementWe recognize with appreciation the theater, labour ward andpostnatalteamforsupportandcaretothepatient’scare.

Informed ConsentWritten informed consent was obtained from the patient’srelativeforpublicationofthiscasereportandanyaccompanyingimages.PermissionwassoughtandgrantedfromMoiTeaching&ReferralHospital(MTRH)priortopublicationofthiscasereport.

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