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InternationalJournalofSurgeryCaseReports5(2014)513–515ContentslistsavailableatScienceDirect
International
Journal
of
Surgery
Case
Reports
j o u r n al ho m e p a g e :w w w . c a s e r e p o r t s . c o m
Totally
inverted
cervix
due
to
a
huge
prolapsed
cervical
myoma
simulating
chronic
non-puerperal
uterine
inversion
夽
Nilgun
Turhan
a,
Serap
Simavli
b,c,
Ikbal
Kaygusuz
d,
Burcu
Kasap
a,∗aDepartmentofObstetricsandGynecology,Mu˘glaSıtkıKoc¸manUniversitySchoolofMedicine,Ankara,Turkey
bDepartmentofObstetricsandGynecology,PamukkaleUniversitySchoolofMedicine,Denizli,Turkey
cLaboratoryofReproductiveEndocrinology,BrighamandWomen’sHospital,HarvardMedicalSchool,Boston,USA
dDepartmentofObstetricsandGynecology,TurgutOzalUniversitySchoolofMedicine,Ankara,Turkey
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t
i
c
l
e
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n
f
o
Articlehistory:
Received13December2013
Accepted14December2013
Availableonline21May2014
Keywords: Cervicalinversion Prolapsedleiomyoma Uterineinversion Vaginalhysterectomy
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INTRODUCTION:Inversionoftheuterusisanextremelyrarecomplicationofthenon-puerperalperiod andiscommonlycausedbybenignsubmucous,especiallyfundal,leiomyomas.Acaseofatotallyinverted cervixduetoaprolapsedhugecervicalleiomyomamimickingchronicnon-puerperaluterineinversion inaperimenopausalwomanispresented.
PRESENTATIONOFCASE:A52-year-oldperimenopausalwomanwasadmittedtoourclinicwithan ulcer-ated,necrotic,infectedandswollenprolapsedmass.Gynecologichistoryrevealedthatshewasadvised myomectomybecauseofhercervicalmyoma2yearsagobutsherefusedtohaveanoperationasshe believedthatherpositivethoughtswouldshrinkthemyoma.Presumeddiagnosisbeforesurgerywas chronicnon-puerperaluterineinversion.Anintraoperativediagnosiswastotallyinvertedcervixdueto ahugecervicalleiomyoma.Vaginalhysterectomywithoutadnexectomy,wasperformed.
CONCLUSION:Thisisthefirstcaseintheliteraturewhichatotallyinvertedcervixduetoaprolapsedhuge cervicalleiomyoma.Cervicalfibroidscangrowinperimenopausalperiodandinextremelyrarecasescan causetotalcervicalinversion.
©2014TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Allrightsreserved.
1. Introduction
Uterinefibroidsarethemostcommontumorsofuteruswhich developin20–40%ofreproductiveagewomen,butcervical leiomy-omasarelessthan5%ofallleiomyomas.1 Pedunculateduterine myomasorsubmucosalcervicalmyomasmayprotrudethrough thecervicalcanalandintothevaginaandmaybecomenecrotic and occasionallyinfected due toinadequateblood supply.2,3 In some cases, theycan cause uterine inversion. Inversion of the uterusis a veryrare complication of thenon-puerperal period andiscommonlycausedbybenignsubmucous,especiallyfundal, leiomyomas.4
Herein,wepresenta huge,necrotic,ulceratedand prolapsed cervicalleiomyomacausinginversionofthecervixandmimicking non-puerperaluterineinversion.Toourknowledgethisisthefirst caseintheliteraturewhichatotallyinvertedcervixduetoa pro-lapsedhugecervicalleiomyomamimickingchronicnon-puerperal uterineinversioninaperimenopausalwoman.
夽 Thisisanopen-accessarticledistributedunderthetermsoftheCreative
Com-monsAttribution-NonCommercial-NoDerivativeWorksLicense,whichpermits
non-commercialuse,distribution,andreproductioninanymedium,providedthe
originalauthorandsourcearecredited.
∗ Correspondingauthorat:DepartmentofObstetricsandGynecology,Mu˘glaSıtkı
Koc¸manUniversity,MedicalSchool,Mu˘gla,Turkey.Tel.:+905052711056;
fax:+902239280.
E-mailaddress:burcuharmandar@gmail.com(B.Kasap).
2. Casepresentation
A52year-oldwoman,gravida4,parity3,abortus1,was admit-tedtoourclinicwiththefollowingcomplaints:lowerabdominal painandamassprotrudingfromthevulvafor6months.Shehad anabdominalmyomectomy14yearsagoandreceivedthreeunits ofblood.Acervicalmyomauteri5cmindiameterwasdiagnosed2 yearsagoinaprivatehospitalandsurgerywasrecommended.She refusedoperationatthattime,believingthatherpositivethoughts wouldshrinkthemass.Shehadninemonthshistoryofasensation ofsomethingcomingoutpervaginum,withblood-stained leuc-orrhoea.Sixmonthsagoaprotrusiondevelopedfromthevagina withseverepain.Shedidnotgotoanyhospitalbecauseshestill believedthatthemasswoulddisappearwithherpositivethinking. Beforeadmissiontoourclinicshetriedherbaltreatmentmethods and continued tothink positivelywith thehopethat her gen-italmass wouldshrink. Aftersix months,shedecidedto apply toour hospital whenthe mass becameulcerated, infected and sensitive.
Generalexaminationwasunremarkable,pulseratewas94beats perminute,bloodpressurewas140/90mmHg,andrespiratoryrate was21cyclesperminute.Theabdominalexaminationwas nor-mal.A13cm×8cmsolid,inflammated,ulcerated,necroticmass, protrudingfromthevaginal introituswasremarkableonpelvic examinationandthebodyofthecervixcouldnotbevisualized sep-aratefromthemass.Despitethefactthatshewasperimenopausal period, her leiomyoma grew from 5cm to13cm in two years
2210-2612/$–seefrontmatter©2014TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Allrightsreserved.
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514 N.Turhanetal./InternationalJournalofSurgeryCaseReports5(2014)513–515
Fig.1. Prolapsednecrotic,ulceratedcervicalleiomyoma,(a)necroticmass,(b) invertedcervix’rugae.
period.Transvaginalultrasonographicexaminationcouldnotbe performedbecauseoftheunreducible,protrudedsensitivevulvar mass.Additionally, theuteruscouldnotrecorded duringpelvic ultrasonography. Preoperative presumed clinical diagnosis was chronicnon-puerperalinversionoftheuterusduetosubmucous leiomyoma.Patientdesiredhysterectomywithoutadnexectomy andavaginalhysterectomywasscheduled.
Pelvicexaminationunderanesthesia, revealedamassarising fromadilatedinvertedcervix,andcervicalrugaewerecovering theexternalsurfaceofthemass(Fig.1).Onbimanualexamination theuterusand adnexeswerepalpated.Intraoperativediagnosis changedtoatotallyinvertedcervixduetoahugeprolapsed cervi-calmyoma(Fig.2).Hysterectomywasbegunbythevaginalroute. Theanteriordissectionwascarriedoutandtheuterovesicalfold opened. Theuterine vesselswere clamped, cut,and transfixed, thenposteriorcul-de-sacwasopened.Uterinecorpusandfundus werenormalinappearance.Thecornualstructuresweredivided
Fig.2.Cervicalinversionduetoprolapsedmyomaswithnormaluterus,(a)uterus,
(b)externalcervicalos,(c)invertedcervix,and(d)leiomyoma.
Fig.3. Normalappearanceofuteruswithprolapsedcervicalmyoma,(a)normal
myometriunanduterinecavity,(b)invertedcervix,and(c)cervicalmyoma.
andsecuredtocompletethehysterectomyvaginally.Theovaries appearednormalandwerenotremoved.Macroscopic examina-tionoftheoperativespecimenconfirmedthediagnosisofatotally invertedcervixduetoahugeprolapsedcervicalleiomyoma(Fig.3). Histopathologicalexaminationofthespecimenconfirmedthe diag-nosisof asubmucousleiomyoma arisingfromthecervixofthe uterus.Therewerenocomplicationsinthepostoperativeperiod andthepatientwasabletobedischargedonthethirdpostoperative day.
3. Discussion
Cervicalleiomyomasareextremely rareandtheincidenceis reportedatabout0.6%intotalhysterectomyspecimens.5Cervical leiomyomascanbecategorizedasextracervicaltype(subserosal location) and intracervical type (occur within the cervix). The complications of cervical leiomyomas include pressure effects onthebladder orurethra,degenerative phenomena, intermen-strualbleeding,pain(pelviccramping),prolapsewithinfectionand torsion.6Whenwesearchedtheliteraturewedidnotseeany inci-dentsofreportedcervicalinversionduetocervicalleiomyoma.To ourknowledgethisisthefirstcaseofatotallyinvertedcervixdue toahugeprolapsedcervicalmyomasimulatinguterineinversion.
Uterineinversionisararecomplicationofthepuerperiumand anon-puerperalinversionisanextremelyrareoccurrence. Usu-ally,non-puerperaluterineinversionpresentsafter45yearsand ismostly related tobenignmyomasand rarelyassociated with malignancies.7 Themechanismoftumor-relatedinversionisnot clearbutitisthoughttobeduetothedistensionandemptying oftheuterinecavity,thinuterinewalls,fundiclocalizationofthe tumor,theexpulsivecontractionsoftheuterus,andtheweightof thetumoritself.8,9
Based on the literature search only one cervical inversion was found. Sivasuriya et al.10 report a case of inversion of a partlyeffacedanddilatedcervixintotheloweruterinesegment at the time of cesarean section. This case was an example of puerperalcervicalinversion,butourcasediffersinthatourpatient
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displayed non-puerperal cervical inversion due to prolapsed cervicalleiomyomagrowingduringtheperimenopausalperiod.
Uterineinversionissuspectedwhengynecologicexamination detectsaprotrudingmassinthevaginaorvulvaandtheuterine funduscannotbepalpatedbybimanualexamination.The deter-miningfindingsareanimpalpablefundusandaninvisiblecervix.11 Clinicaldiagnosisofchronicnon-puerperaluterineinversionis dif-ficult,especiallyifvaginalexaminationcannotbeperformed.Inour case,therewasaprotrudingmassinthevulva,thecervixcould notbevisualizedseparatelyfromthemassandtheuterinefundus couldnotbepalpated.Thesefindingsmadeusstronglyconsider thepossibilityofnon-puerperaluterineinversion.
Ultrasonography mightbe a usefultool for diagnosis. Sono-graphic characteristics of complete uterine inversion are a ‘U’-shapeduterinecavityinthelongitudinalplane.12Wecouldnot performtransvaginalUSGandunfortunatelypelvic ultrasonogra-phydidnotprovideanyadditionalinformation.Examinationdone underanesthesiarevealedaninvertedcervixcoveringthemassand afundusbybimanualpalpationandtheintraoperativediagnosis waschangedtoatotallyinvertedcervixduetoahugeprolapsed cervicalleiomyomasimulatinguterineinversion.
Prolapsed leiomyoma treatment is vaginal myomectomy or hysterectomy.Ourpatientdesiredhysterectomywithout adnex-ectomyandvaginalhysterectomywasperformedandthepatient’s postoperativecoursewasuneventful.
Wereportacaseoftotallyinvertedcervixduetoahugecervical leiomyomamimickingnon-puerperaluterineinversionina peri-menopausalwoman.Cervicalfibroidscangrowinperimenopausal periodandinrarecasescancausetotalcervicalinversion.
Conflictofinterest
None.
Funding
None.
Ethicalapproval
Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonrequest.
Authorcontributions
NilgunOzturkTurhan,SerapSimavlı,andIkbalKaygusuzhelped for study design, study collection, and writing. Burcu Kasap involvedinwriting.
References
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8.LascaridesE,CohenM.Surgicalmanagementofnonpuerperalinversionofthe uterus.ObstetGynecol1968;32:376–81.
9.KrenningRA,DorrPJ,deGrootWH,deGoeyWB.Non-puerperaluterine inver-sion.Casereport.BrJObstetGynaecol1982;89:247–9.
10.SivasuriyaM,HerathHP.Inversionofcervixuteriatcaesareansection.BrMedJ 1976;1:746–7.
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