A case of 20-week abortion in a rare communicating rudimentary horn of a misinterpreted unicornuate uterus, incorrectly diagnosed as bicornuate: A serious hazard!

A

case

of

20-week

abortion

in

a

rare

communicating

rudimentary

horn

of

a

misinterpreted

unicornuate

uterus,

incorrectly

diagnosed

as

bicornuate:

A

serious

hazard!

Luigi

Della

Corte

*

,

Annamaria

Fabozzi

,

Pierluigi

Giampaolino

,

Gabriele

Saccone

,

Laura

Micol

Pizzuti

,

Valeria

Romeo

,

Simone

Maurea

,

Giuseppe

Bifulco

a

DepartmentofNeuroscience,ReproductiveSciencesandDentistry,SchoolofMedicine,UniversityofNaplesFedericoII,Naples,Italy

b

DepartmentofPublicHealth,UniversityofNaplesFedericoII,Naples,Italy

c

IstitutodiBiostruttureeBioimmagini(IBB-CNR),Naples,Italy

d

DepartmentofAdvancedBiomedicalSciences,UniversityofNaplesFedericoII,Naples,Italy

ARTICLE INFO

Articlehistory:

Received29December2018

Receivedinrevisedform30January2019

Accepted17February2019

Availableonlinexxx

Keywords:

Communicatingrudimentaryhorn

Magneticresonanceimaging

Laparotomy

Second-trimesterabortion

Uterinemalformation

ABSTRACT

Femalegenitalmalformations,astheunicornuateuterus,aredeviationsfromnormalanatomythatcould impair thereproductivepotential ofawomanorherhealth.We presentararecaseofa20-week spontaneousabortionina24yearsoldpatientaffectedbyamisunderstoodunicornuateuteruswith communicatingrudimentaryhorn,previouslydiagnosedasbicornuate,andforthisreasonsubjectedto inductionofabortivelabor,usingmifepristoneandgemeprost.FollowingtheultrasoundexamandMRI, performedduetothefailureoftheabortiveprocedure,revealedthediagnosisofunicornuateuteruswith (notclear)communicatingaccessoryhornpregnancy,thentreatedwithlaparotomy. 3D-ultrasonogra-phy,andaboveallMRI,shouldbeperformedinallthosecasesofsuspecteduterineanomalies,especially inpresenceofpregnancyorabortion,withtheaimofavoidingwrongtreatments,whichleadstoahigh riskofuterine rupture.Inthis case,giventheuncertaintyofimagingexamsperformedin suchan advancedsecond trimesterofpregnancy,onlythesurgical approachwas ableto discoverthereal communication.

©2019PublishedbyElsevierB.V.

DearEditor,

Femalegenital malformations,astheunicornuateuterus,are deviationsfromnormalanatomythatcouldimpairthe reproduc-tivepotentialofawomanor,incomplexcases,woman’shealth[1]. Theincidenceofrudimentaryhornpregnancyisreportedtobeone in76,000pregnancies[2],andin75_–83%ofcasesthepregnancy developsinnon-communicatingrudimentaryhorn[3].

Here,wepresentararecaseofa20-weekspontaneousabortion ina24yearsoldpatientaffectedbyamisunderstoodunicornuate uteruswithcommunicatingrudimentaryhorn, previously diag-nosedasbicornuate,anditsclinicalandsurgicalmanagement.

A24-year-oldwoman,primigravidaat20weeksofgestation, wasadmittedtoourhospitalwiththediagnosisofabortion.She reportedhavingreceiveddiagnosisofbicornuateuterusabout1

yearbefore,bothby3D-transvaginalultrasoundandhysteroscopy, performedbyhergynecologistfromwhomrelativedocumentation wasprovided.Atadmission,anofficeultrasoundwasperformed, confirmingthepresenceoftwohorns,inoneofthemadeadfetus with20weeksbiometrywas found.Atvaginalexaminationthe presenceofasinglecervixwasdeterminedandnovaginalbleeding wasobserved.Parameterswerenormal;therefore,accordingtothe protocolofourDepartment,pharmocologicalinductionofabortive laborwasundertakenwithMifepristone600mgperos,followed byvaginaladministrationofgemeprost,onevaginalsuppository everythreehoursforatotalnumberof5suppositories.Despitethe onsetofuterinecontractions,thecervixwasstillunaltered.Dueto failureof induction,doubtaroseconcerningthetypeofuterine malformation.Ourskilledsonographerperformeda3Dultrasound examinationusingaFoleycatheterplacedinthecervix,withthe aimofassessingtherealcommunicationbetweentheuterusand thehorncontainingthepregnancy.Ultrasoundscanssuggesteda continuitybetweencervicalcanalandtheright,emptyhemiutero, with a thickened endometrium as a decidual reaction. The pregnancy appeared in theother hornwithout communication withthecervix.Thepatient’sconditionwasstableandtherewere

* Correspondingauthorat:DepartmentofNeuroscience,ReproductiveSciences

andDentistry,SchoolofMedicine,UniversityofNaples“FedericoII”,ViaPansini5,

Naples,Italy.

E-mailaddress:dellacorte.luigi25@gmail.com(L.DellaCorte).

https://doi.org/10.1016/j.ejogrb.2019.02.018

0301-2115/©2019PublishedbyElsevierB.V.

EuropeanJournalofObstetrics&GynecologyandReproductiveBiologyxxx(2019)xxx–xxx

GModel

EURO10729No.ofPages3

Pleasecitethisarticleinpress as: L.Della Corte,etal., Acaseof 20-week abortioninarare communicating rudimentaryhornof a misinterpretedunicornuateuterus,incorrectlydiagnosedasbicornuate:Aserioushazard!,Eur JObstetGynecol(2019),https://doi.org/ 10.1016/j.ejogrb.2019.02.018

ContentslistsavailableatScienceDirect

European

Journal

of

Obstetrics

&

Gynecology

and

Reproductive

Biology

no ultrasound or clinical signs of uterine rupture. Magnetic resonance imaging (MRI) examination without contrast agent injectionconfirmedthepresenceoftwouterinehornsofwhich one, anteriorly located and containing a dead fetus, did not demonstrate a clear communication with the cervical canal (Fig. 1A–C). A diagnostic laparoscopy was carried out and it showedabigrudimentaryuterinehornwithpregnancyinside.For thisreason,wedecidedonalaparotomicapproach(Fig.1D–F):the rudimentaryhornwasexcised andtheleftroundligamentwas sutured tothe uterusrestoring the uterineanatomy. Afterthe removalof therudimentary horn, a small communication was surprisinglyidentifiedwiththeisthmusofdominantuterus,that wassoonsutured.Thedominantuteruswassuspendedattheleft roundligamentandtheabdomenclosedfollowinghemostasis.The patientwasdischargedinasatisfactoryconditiononthefifthday afterkidneyanomalieswereruledout.

A unicornuate uterus communicating withthe contralateral uterinecavityis a ClassIVa anomalyaccording toESHRE/ESGE Consensus[2].Itaccountsfor2.4e13%ofMulleriananomalies.A solitaryuterinehorncanbeobservedinupto35%ofpatients[4].In 66%ofcasesthereisnocommunicationbetweentherudimentary hornandprimaryhorn.

Hornruptureoccursin80–90%ofthesepregnanciesduringthe secondtrimesterand10%ofthepregnanciesproceedtoterm,but only 2% reach fetal survival. An early diagnosis and a proper managementaremandatory.

Tsafrir et al. suggest criteria for the early diagnosis of rudimentary horn pregnancy: (1) pseudo pattern of an asymmetrical bicornuate uterus (2) absent visual continuity betweenthecervicalcanalandthelumenofthepregnanthorn, and (3) the presence of myometrial tissue surrounding the gestationalsac[5].

Thetransvaginalultrasonographydetectionratecoulddendsto displacethecontralateralhalfoftheuterus,makingitdifficultto demonstrateuterineanomaly.MRIhasbecomethegoldstandard fortheevaluationofcongenitaluterineanomalies.

The management consists of excision of the pregnant rudimentary horn and ipsilateral fallopian tube, traditionally bylaparotomy.

In this case of an ongoing abortion in the late second trimester, only laparotomy was able to detect the clear communicationbetweentheunilateraluterusandthe contra-lateral uterine cavity. Nevertheless, 3D-ultrasonography, and above all MRI, should be performed early in all cases of suspected uterine anomalies, especially in the presence of pregnancy or abortion, with the aim of avoiding erroneous treatments,leadingtoahighriskofuterinerupture,fetaldeath and/ormaternalmorbidityandserioushazard.

Conflictofinterest

Theauthorsdeclarethattheyhavenothingtodisclose.

Fig.1.A–C.CoronalTSET2-weighted(A),sagittalTSET2-weightedwithfatsuppression(B).OntheaxialTSET2-weighted(C)images.Arudimentaryhorncontaininga

20-weeksdeadfetusisdetectable(blackasterisksinAandB);thedominantuterusisposteriorlylocated(whitearrowinB).Onaxialplane(C),aclearcommunicationwiththe

cervicalcanal(whitearrow)isdetectableonlyforthedominantuterus(whiteasterisk).D.Therudimentaryuterinehornwithpregnancyinside.E.Topviewofrudimentary

uterinehornwithoutpregnancy.F.Thedominantuterusaftertheremovalofrudimentaryuterinehornandthesutureoftheleftside-wall.

2 L.DellaCorteetal./EuropeanJournalofObstetrics&GynecologyandReproductiveBiologyxxx(2019)xxx–xxx

GModel

EURO10729No.ofPages3

Please citethis articleinpress as:L.Della Corte,et al., Acaseof 20-weekabortionina rarecommunicatingrudimentary hornof a misinterpreted unicornuateuterus,incorrectlydiagnosedasbicornuate:Aserioushazard!,EurJObstetGynecol(2019),https://doi.org/ 10.1016/j.ejogrb.2019.02.018

Financialsupport

Nofinancialsupportwasreceivedforthisstudy. References

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[2]GrimbizisGF,GordtsS,DiSpiezioSardoA,BruckerS,DeAngelisC,GergoletM, etal.TheESHRE/ESGEconsensusontheclassificationoffemalegenitaltract congenitalanomalies.HumReprod2013;28(August(8)):2032–44.

[3]JohansenK.Pregnancyinarudimentaryhorn.ObstetGynecol1983;61:565e7. [4]OlpinJD,MoeniA,WillmoreRJ,HeilbrunME.MRimagingofMüllerianfusion

anomalies.MagnResonImagingClinNAm2017;25(August(3)):563–75. [5]TsafrirA,RojanskyN,SelaHY,GomoriJM,NadjariM.Rudimentaryhorn

pregnancy:first-trimesterprerupturesonographicdiagnosisand confirma-tionbymagneticresonanceimaging. JUltrasoundMed2005;24(February (2)):219–23.

L.DellaCorteetal./EuropeanJournalofObstetrics&GynecologyandReproductiveBiologyxxx(2019)xxx–xxx 3

GModel

EURO10729No.ofPages3

Pleasecitethisarticleinpress as: L.Della Corte,etal., Acaseof 20-week abortioninarare communicating rudimentaryhornof a misinterpretedunicornuateuterus,incorrectlydiagnosedasbicornuate:Aserioushazard!,Eur JObstetGynecol(2019),https://doi.org/ 10.1016/j.ejogrb.2019.02.018