Endometrial Adenocarcinoma Recurrence Presenting With Tibial Metastasis: Report of a Case

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InternationalJournalofSurgeryCaseReports36(2017)15–17

ContentslistsavailableatScienceDirect

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

Endometrial

adenocarcinoma

recurrence

presenting

with

tibial

metastasis:

Report

of

a

case

Mehmet

Salih

Söylemez

_,

_Bahattin

_Kemah

_,

_Umut

_Perc¸

_em

_Orhan

_Söylemez

_,

Bülent

Kılıc¸

_,

_Korhan

_Ozkan

a_{BingolStateHospital,DepartmentofOrthopaedics&Traumatology,Bingol,Turkey} b_{AgriStateHospital,DepartmentofOrthopaedics&Traumatology,Agri,Turkey} c_{BingolStateHospital,DepartmentofRadiology,Bingol,Turkey}

d_{I.GelisimUniversitySchoolofHealthSciences,Istanbul,Turkey}

e_{I.MedeniyetUniversityGöztepeTrainingandResearchHospital,OrthopaedicsandTraumatologyDepartment,Istanbul,Turkey}

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Articlehistory:

Received14January2017

Receivedinrevisedform16April2017 Accepted16April2017

Availableonline18April2017 Keywords:

Casereport Orthopaediconcology Bonemetastasis Solitarybonelesion Endometrialadenocarcinoma

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INTRODUCTION:Metastaticbonediseaseatextremitiesismostlyassociatedwithlung,liver,prostat, thyriodorbreastmalignancies.Thereforsurgeonsgenerallytendstoseekforaprimarytumor origi-natingfromtheseorgans.Hereinacaseofendometrialadenocarcinomarecurrencethatpresentedwith symptomsoftibialpainisdescribed.

PRESENTATIONOFCASE:59year-oldwomanwasadmittedtoourorthopaediconcologyunitwithpain, swellingandtendernessatrightcrurisfortwoweekswithoutanytraumahistory.Hermedical his-toryrevealedthatshehadatotalabdominalhysterectomyandbilateralsalpingo-oophorectomy.During follow-upsnorecurrencehadbeendetected.InitialX-raysoftherighttibiashowedalyticandexpansile masslocatedattheshaftofthetibiasuggestingmetastasis.Awideresectionofthelesionwithclear marginswasperformedtwoweeksafterfirstadmittance.Resectedareawasreplacedbyfreshfrozen femoralshaftallograft.Atpostoperative17thmonth.X-raysobtainedatlastfollow-updemostratedfull healingandintegrationofallograft.

DISCUSSION:Endometrialadenocarcinomaisadiseaseofpostmenapousalwomenwith95%ofthecases occurringaftertheageof40years.Patientswithadvancedorrecurrentendometrialcanceroftenhave distantmetastasesfoundwithinthelymphnodes,liver,and/orlung.

CONCLUSION:Recurrenceofendometrialcancerasasolitarybonelesionisararesituation.Wideresection andreconstructionwithanallograftoranintercalarprosthesismightbeanoptiontoincreasesurvival andpossiblecureofthepatient.

©2017TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction

Endometrialadenocarcinomaisoneofthegynecological malig-nancieswhichtendstooccurinpostmenopausalwomen.Irregular uterinebleedingisthemostcommonclinicalpresentationofthese adenocarcinomas.Malignanciesoriginatingfromuterinetissuecan metastasetoseveralorganswithapredilectiontolungsandliver. Metastasistoskeletalsystemisextremelyrareandmostlyhave beenreportedtobeinaxialskeleton[1].

Metastaticbonediseaseatextremitiesismostlyassociatedwith lung,liver,prostat,thyriodorbreastmalignancies.Therefor

sur-∗ Correspondingauthor.

E-mailaddresses:slhsylmz@gmail.com(M.S.Söylemez),

bahattinkemah.md@gmail.com(B.Kemah),umutpercem@gmail.com

(U.P.O.Söylemez),dr.buletk@hotmail.com(B.Kılıc¸),korhanozkan76@gmail.com

(K.Ozkan).

geonsgenerallytendstoseekforaprimarytumororiginatingfrom theseorgans[1,2].Howeveralthoughitisveryrare,solitarybone lesionshasbeenreportedtobeevenfirstpresentationsiteofa previouslycured endometrialadenocarcinoma.Hereina caseof endometrialadenocarcinomarecurrencethatadmittedtoour uni-versityhospitalwithsymptomsoftibialpainisdescribed. 2. Presentationofcase

59year-oldTurkishwomanwasadmittedtoourorthopaedic oncologyunitwithpain,swellingandtendernessatrightcrurisfor twoweekswithoutanytraumahistory.Shehadnoanyother com-plaintandshehadwalkedintotheclinic.Shedidnothadahistoryof hereditarygeneticdisorderandshewasnotsmoking.Hermedical historyrevealedthatshehadatotalabdominalhysterectomyand bilateralsalpingo-oophorectomywiththediagnosisofendometrial adenocarcinomafollowedbychemotherapytwoyearsago.During follow-upsnorecurrencehadbeendetected.InitialX-raysofthe

http://dx.doi.org/10.1016/j.ijscr.2017.04.009

2210-2612/©2017TheAuthor(s).PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://

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16 M.S.Söylemezetal./InternationalJournalofSurgeryCaseReports36(2017)15–17

Fig.1.Preoperativeanterior-posteriorandlateralX-raysshowinglyticexpansile

masslocatedattheshaftofthetibia.

Fig.2. Anterior-posteriorandlateralX-raysshowingtibiahealedatpostoperative

firstyear.

righttibiashoweda 3×1,5cmlyticandexpansilemasslocated attheshaftofthetibiasuggestingmetastasis(Fig.1).MRIimages demonstratedcontrastenhanced3_×1,5cmmedullaryexpansile lesionsurroundedbyboneedema.AwholebodyPET/CTwas per-fomedto detect any other possible lesions. HowevererPET/CT imagingdidn’tdemonstrateanyincreasedFDGuptakeatanyother location.Atrue-cutbiopsywasundertakenwhichrevealedan ade-nocarcinomaconsistentwithaprimaryendometrialtumor.

A wide resectionof the lesion withclear marginswas per-formed two weeks after first admittance. Resected area was replacedbyfreshfrozenfemoralshaftallograftandwasfixedwith intramedullarynailandplate.Osteotomylineswassupportedby impactingautogreftobtainedfromiliaccrest.Thepatient under-went additional adjuvant chemotherapy. At postoperative 3th. monthweightbearingwasallowedandnoreccurencewasdetected atpostoperative 17thmonth.X-rays obtainedat last follow-up demostratedfullhealingandintegrationofallograft(Fig.2). 3. Discussion

Metastaticbone diseaseis themost commonmalignancy of bone.The mostcommonprimarycancersite is thelung (50%), followedbythecolon,breastandgenito-urinarytract[2]. Endome-trialadenocarcinomaisadiseaseofpostmenapousalwomenwith 95%ofthecasesoccurringaftertheageof40years[3].Patients withadvanced orrecurrent endometrialcanceroftenhave dis-tantmetastasesfoundwithinthelymphnodes,liver,and/orlung [3,4].However,there havebeenonlyfewcasesreportedof

pri-maryendometrialcancerwithmetastasistothebone.Ithasbeen reportedtovary2–6%[5].Inanautopsystudy,the correspond-ingfrequencywas25–75%[6].Bonemetastasisaremostlyseen asrecurrenceandsurvivalisbetterwhencomparedtothosewho sustainedsolitarybonemetastasisatprimarydiagnosis[1,7].

Acralmetastases(matastasesdistaltotheelbowandtheknee) afterendometrialadenocarcinomaisveryrare.Bonemetastasesare mostlyseeninvertebralcolumnandpelvicringbecauseofbatson venousplexuswhich drainsperiuterinandparavertebralregion [3,5,6,8]. However, in rare cases metastasisto talus, calcaneus, tarsusandfemurhadalsobeenreported[4].Endometrial adeno-carcinomapresentingwithabonemetastasisatthefirstdiagnosis israrelyseen.Usuallyfirstsignisabnormaluterinebleedingand pelvicpain,yetendometrialadenocarcinomadiagnosedaftertibial metastaseshavebeenreportedin7casesuptonow[1,3,9–13]. Metastatic bone disease is usually seen after first diagnosis of endometrialadenocarcinomaanddurationbetweendiagnosisand metastasisis6monthsto5years[6].Howeverrecurrenceofthe diseasepresentedwithisolatedtibialmetastasisaftertreatmentis veryrareandthisis5th.casethathavebeenreporteduptonowin theenglishspeakingliterature[8,11,14].

Isolatedbonemetastasisafterendometrialadenocarcinomais seenin50%ofthepatientsthathaveabonemetastasisandthese metastasesaremostlywithlowgradediseaseashighgradedisease tendstobemulticentric[8].Treatmentofmetastaticbonetumors dependsonstagingandtumorextent.Amputativesurgeryforacral metastasesmaybeincluded,forcancerswithpoorresponseto radi-ationtherapyand chemotherapy[15].Painfulmetastasestothe fingersofbothhandsandfeetshavebeenadvisedtobetreatedwith amputationtoincreaselifecomfortduringtheterminalperiodand toeasepatient’sdailylifeactivities[16,14].Howeverasitwasseen inourcaseifdiseasehavebeenpresentedwithanisolatedsolitary bonemetastasisasurgicalproceduremaybeeffectivetoobtaina longerlifeexpectancywhileprotectingthelimbandpossiblecure ofthedisease.Althoughsurvivalfollowingmetastaticbonedisease havebeenreportedtobedismal,ourpatientiswellafter17months postoperativelywithnorecurrence[2,10].Wideresectionthatis tumoraltissueremovedwithamarginofnormal tissuemaybe theprimarygoalinpatientswithisolatedmetastasesof endome-trialcancertocontrolthediseaseprogression.Ourpatientwasvery satisfiedwithheroperationasshehadnorestrictioninherlifeand wascuredduringthatfollow-uptime.

4. Conclusion

Recurrenceofendometrialcancerasasolitarybonelesionis araresituation.Althoughsolitarybonelesionsofextremitiesare mostlyassociatedwithlung,liver,prostat,thyriodorbreast malig-nancies,endometrialcancershouldbekeptinmindfordifferential diagnosisaswell.Wideresectionandreconstructionwithan allo-graftoran intercalarprosthesismightbeanoption toincrease survivalandpossiblecureofthepatientasseeninourcase. Conflictofinterest

Theauthorsdeclarethattheyhavenocompetinginterests. Sourceoffunding

Anystudysponsorshadnoinvolvement. Ethicalapproval

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Asthese

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M.S.Söylemezetal./InternationalJournalofSurgeryCaseReports36(2017)15–17 17

wasareportofaninterestingcaseandwasnotatrailoran obser-vationalresearchtherewasnoneedforanethicalapproval.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournal.

Authorscontribution

KorhanOzkan,BülentKilic¸,andUmutPerc¸emOrhanSöylemez contributedtotheconceptionanddesignofthestudy,carriedout theliterature research,manuscript preparation and manuscript review.

MehmetSalih Soylemezand BahattinKemah, wereinvolved withthecaseandwritingofthemanuscript,generalmanagement ofthepatientandrevisedthemanuscriptforimportantintellectual content.

Allauthorsreadandapprovedthefinalmanuscript.

Registrationofresearchstudies

Nameoftheregistry:EndometrialAdenocarcinomaRecurrence PresentingWithTibialMetastasis:ReportofACase.

Uniqueidentifyingnumber(UIN):researchregistry2387.

Guarantor

Assoc.Prof.Dr.KorhanOzkan. MehmetSalihSöylemez,MD.

Acknowledgement

Thismanuscripthasbeenpreparedandreportedinlinewith SCAREcriteria[17].Allauthorsdeclarethattheyhavenoconflict ofinterest.

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