case report - iKnow

(1)

ContentslistsavailableatScienceDirect

International Journal of Surgery Case Reports

jo u r n al ho me p a g e :w w w . c a s e r e p o r t s . c o m

Reconstruction of osteosarcoma of the proximal tibia using bone on polyethylene hemiarthroplasty knee joint system: A case report

Yogi Prabowo

^a

, Muhammad Rizqi Adhi Primaputra

^a,∗

, Evelina Kodrat

^b

aDepartmentofOrthopaedic&Traumatology,CiptoMangunkusumoNationalCentralHospitalandFacultyofMedicine,UniversitasIndonesia,Jalan DiponegoroNo.71,CentralJakarta,Jakarta10430,Indonesia

bDepartmentofAnatomicalPathology,CiptoMangunkusumoNationalCentralHospitalandFacultyofMedicine,UniversitasIndonesia,JalanDiponegoro No.71,CentralJakarta,Jakarta10430,Indonesia

a r t i c l e i n f o

Articlehistory:

Received20February2020

Receivedinrevisedform12May2020 Accepted13May2020

Availableonline21May2020

Keywords:

Osteosarcoma Reconstruction Polyethylene Hemiarthroplasty

a b s t r a c t

INTRODUCTION:Currenttreatmentintreatingpatientwithosteosarcomaiscombinationofchemotherapy andsurgery,eitherlimb-sparingorlimb-ablationsurgery.Onechallengeinlimb-sparingtechniquein childrenishowtodealwiththeremaininggrowthofthebone.Wecreatedalimb-sparingreconstruction techniquethatcanbeﬁttedinmanytypesofhospitalandhaveagoodfunctionaloutcome.

CASEPRESENTATION:Wereportedacaseof13-years-oldfemalewithleftkneepainsince8months priorhospitaladmissionwithprevioustreatmenttoabonesetterandopenbiopsyatprevioushospital.

Histopathologicalﬁndingsdemonstratedosteosarcoma.Thepatienthadaneoadjuvantchemotherapy andfollowedwithlimb-sparingsurgery.Weperformedwideexcisionofproximaltibiaandreconstruc- tionusingaboneonpolyethylenehemiarthroplastysystemofthekneejoint.Patientthencontinuedon adjuvantchemotherapy.Lateron,localrecurrenceoccurredandweperformedexcisionagain.Within oneyear,thepatientcouldwalkfullweightbearing,performdailyactivitieswithnolimitationandno pain,andshehadMSTSscoreof21.

DISCUSSION:Limb-sparingsurgeryremainsachallengingprocedureinskeletallyimmaturepatients.This boneonpolyethylenetechniquewouldbefunctionalduetohighadaptabilityinpaediatricpatientsin ordertodecreasethenumberofsurgeriesuntiltheﬁnalgoaloflimbequalization.

CONCLUSION:Boneonpolyethylenehemiarthroplastysystemenablesgoodandreliablefunctionalout- comewhilemaintainingthekneejointfordailyactivity.Itcanbechosenasoneofviableoptionsin treatingosteosarcomaaroundthekneejointinchildren.

1. Introduction

Osteosarcomais themostcommonprimary malignant bone tumorinchildren[1].Themostcommonsiteisdistalfemur,fol- lowedbyproximaltibia[2,3].Currenttreatmentintreatingpatient withosteosarcomais combinationofneoadjuvantandadjuvant chemotherapyand surgery,eitherlimb-sparingorlimb-ablation surgery.Oneofthechallengeinlimb-sparingtechniqueinchildren ishowtodealwithremaininggrowthofthebone[4,5].Oneof thetechniquesforlimb-sparingreconstructionaroundthekneeto solvetheproblemisendoprosthesesreplacementusingmodular type[4,6].However,thismodularendoprosthesestypecannotbe doneinourcountryduetoitshighcost.Thereforewecreateda limb-sparingreconstructiontechniquethatcanbeﬁttedinmany

∗ Correspondingauthorat:DepartmentofOrthopaedic&Traumatology,Cipto MangunkusumoNationalCentralHospital,JalanDiponegoroNo.71,CentralJakarta, Jakarta10430,Indonesia.

E-mailaddress:[email protected](M.R.A.Primaputra).

typesofhospitalandhaveagoodfunctionaloutcome.Thiswork hasbeenreportedinlinewiththeSCAREcriteria[7].

2. Casepresentation

A13-years-oldfemalepresentedwithpainonherleftkneesince 8monthsago.Thepainwascontinuousandgotworseatnight.The patientwenttoatraditionalmasseuseandwasgiventopicalherbal ointment.Threemonthslater,alumpappearedatherkneeatasize oftabletennisballandgettingbigger.Thepatientthenwenttoa generalsurgeon,andhistopathologicalexaminationdemonstrated bonetumor.Shewassubsequentlyreferredtoourhospital.

Physicalexaminationdemonstratedalumpontheanteriorpart ofproximaltibiawithprevioussurgicalbiopsyscarand venec- tation. The lump had hard consistency with ill-deﬁned border, smoothsurface,immobile,and pain(visualanaloguescale2-3).

Sizecircumferentialofthelegwas33cmcomparedto27cmonthe contralateral.Neurovascularwasgood.Therangeofmovementof bothofthekneeandanklewasnormal(Fig.1).

https://doi.org/10.1016/j.ijscr.2020.05.013

org/licenses/by/4.0/).

(2)

CASE REPORT – OPEN ACCESS

Y.Prabowoetal./InternationalJournalofSurgeryCaseReports72(2020)188–196 189

Fig.1. ClinicalPictureAfterChemotherapy.

Plainradiographysuggestedmixedlyticandblasticbonylesion inmetaphysealareaofproximaltibia,withill-deﬁnedmargin,wide transitionalzone,periostealreaction,matrixosteoidandalsosoft tissueinvolvement(Fig.2a).Plainradiographyofchestshowedno signofpulmonarymetastasis(Fig.2b).Contrast-enhancedmag- neticresonanceimaging(MRI)showedheterogeneousmassandno neurovascularbundleinvolvement(Fig.3).Laboratoryexamination demonstrated increasederythrocyte sediment rate47mm/hour (N:<15),andlactatedehydrogenase717U/L(N:<480).

We dida reviewslide ofthepreviousbiopsysamplebythe pathology of anatomy in our hospital. The result came out an

osteosarcomawithosteoblasticcells.Thenthecasewasbroughtto clinicopathologicalconference(CPC–Aboardconsistingofexperts fromOrthopaedics Surgeon, Radiology,and PathologyAnatomy Department)–andthepatientunderwentchemotherapyneoad- juvantfollowedwithsurgerybylimbsalvagesurgeryprocedure, wideexcisionandreconstructionwithhemiarthroplastysystem.

The patientunderwent chemotherapy neoadjuvantfor 4 cycles weekswithregimentsCisplatinandDoxorubicin.Afterthat,patient underwentalimbsalvagesurgery.

Intraoperatively,wemanagedtoperformwideexcision,while preserving somepartof patellar tendon,medial collateral liga- ment,andcruciateligament(Fig.4).Weexcisedthetumorand thenwemeasuredthebonedefectis19cmfromjointline(Fig.5).

Thenwe reconstructeda hemiarthroplastysystemtofill in the defect.Weusedatibialcomponentandinsertoftheprimarytotal kneereplacementimplantandweconnectedtothedistalusing aKuntscherNailandaproximaltibiallockingplate.Weputthe nailintramedullarytodistalremainingbone(Fig.6).Wefixated alltheimplantswithbonecementandwereconstructedthesoft tissueusingameshinanteriorsideofimplants.Wereconstructed thecruciateligamentthroughtheinsertandtiedtothemeshand alsowetiedallthemedialcollateralligamentandtheremaining patellartendontothemesh(Fig.7).We addedmedial gastroc- nemiusflaptoaddcoverageofsofttissuecoveringtheimplants (Fig.8).Weputaposteriorsplintdirectlyafteroperation(Fig.9).The patientroutinelycontrolledtoouroutpatientcliniceverymonth.

After6weeks,weremovedthesplintandpatientstartedrange ofmotion6weeksafterandpartialweightbearingusingbilateral crutches.Subesquently,weconﬁrmedthehistopathologyresultfor HUVOSresult(Fig.10)andcontinuedwithchemotherapyadjuvant foranother5cycleswithregimentsofiphosphamide,cisplatinand adriamycin.

Eightmonthslater,lumpre-appearedatthesamesiteofpre- viouslesionwithpain.Thelumpsizewas2×2×2cmwithhard consistencyandpainvisualanaloguescale2-3(Fig.11).Novenec- tation,nosinusorwoundappeared.X-rayconﬁrmedthatposition oftheimplantstillgoodwithnobonylesion.Then,wedidexci- sion ofthe massand it came out the resultwas osteosarcoma (Figs.12and13).

On1yearoffollow-up,thepatientcouldwalkwithfullweight bearingwithoutanyadditionalaid.Shecomplainednopain,and

Fig.2.A.Crurisxrayshowedamixedlyticandblasticlesion.B.Thoraxxrayshowednosignofmetastasis.

(3)

Fig.3.MRIshowedaheterogenousmassinepiphysisextendingtodiaphysisoftibia,16cmfromtibiofemoraljoint,withoutneurovascularbundleinvolvement.

Fig.4. A.Patellartendonpreservation.B.Medialcollateralligamentpreservation.

shecoulddoherdailyactivitywell.Theleglengthdiscrepancywas approximately1cm,herkneeﬂexion-extensionrangeofmotion was10 to 50 degrees within stable varus valgus test, motoric strength4,andtheMSTSscorewas21(Fig.14).

3. Discussion

Osteosarcomaisamalignanttumorwhicharisesfromames- enchymalstemcellprecursorthatproducesimmaturewovenbone (osteoid)[1,5].Itisthemostcommonpaediatricbonemalignancy [6].Thepeakincidenceofthismalignancyisbetweentheageof

10and20yearsofage,andtheannualincidenceisapproximately 2to3per106,000individuals[5].Themostfrequentsitesofsuch tumorincludedistalfemur,proximaltibiaandproximalhumerus [2,3].

Inthepast, osteosarcomawastreatedby amputationsolely.

Nowadays, neoadjuvant preoperative and adjuvant chemotherapy regimens exist, and they allow safe limb-sparing resec- tions, thereby improving survival rates [1–3,5,8]. Neoadjuvant chemotherapyhasseveralgoals:1)reducingtumorsize,2)clear- ingmicro-metastasis,and3)facilitatingbettermarginationofthe primarytumorthusallowinglimbsalvagesurgerytobeperformed

(4)

CASE REPORT – OPEN ACCESS

Fig.5. A.Tumorexposed.B.Bonedefect19cmaftertumorremoval.C.Grosspathologyofthetumor.

ifpossible[1].Thetotaldurationofthetreatmentrangedfrom6to 10months;thisdependsonchemotherapyresponse[1,6].Withthe recentavailabilityofmultimodaltreatmentcombiningchemother- apy,andsurgicaltechniques,70–85%ofmalignanttumorscouldbe efﬁcientlytreatedwithlimbsalvage.Withcurrentmodalities,the 5-yearoverallsurvivalandevent-freesurvivalratesachievedwas about70%and58%,respectively.However,thosewhopresentwith metastaticdiseasestillhaveunfavourableoutcomes,withoverall 5-yearsurvivalofapproximately34%[2,5,6,8,9].

Limb-salvagesurgeryprovidesgoodoncologicalandfunctional outcomes,aswellassatisfactorypsychologicalresults[1].Despite numerousreportsregardingthesurgicaltechniques(e.g.allografts andarthrodesis),thetreatmentoptionsforreconstructingboneand soft-tissuedefectsafterresectionremainaseriouschallengefor orthopaedicsurgeons,particularlyinskeletallyimmaturepatients [4,5,9–11].Astheepiphysisofbothsidesofthekneejointaccount formorethantwo-thirdsoflongitudinallowerlimbgrowth,resec- tionofosteosarcomaaroundkneejointsin skeletallyimmature patients presents a concern regarding limb-length discrepancy (LLD)[4,5,9–12].

Withrecentadvancesinprosthesisdesign,modularcemented endoprosthesesareabundantlyusedtoreconstructdistalfemur andproximaltibiadefectsafterbonetumorresection.Theseendo- prosthesesarefrequentlyusedduetotheirlowincidenceofaseptic loosening; moreover, they provide better functional outcomes [1,5,6,9,13].

Theproximaltibiaisthesecondmostcommonsiteofosteosar- coma after distal femur. This procedure is challenging due to variousfactors.Often,complicationsandlossoffunctionfollow-

ingresectionoftheextensormechanismoccur[14,15].Numerous methods are available to improve extensor mechanism; these includedirectreattachmentofthepatellartendontotheprosthesis andattachmentofthepatellartendontothetransposedgastroc- nemiusmuscleﬂap[14,15].Medialgastrocnemiusmuscleﬂapalso showntodecreasetheriskofinfectionandpromotegoodheal- ing[14–16].Allograftswereusedinordertodecreasetheriskof loosening,synovitis,andtrauma[16].

Toavoidthehighcostofmodularendoprosthesisandpreserve kneejointpreservationin children,we designeda combination ofarthroplastyandinternalfixationtoovercomethedefectafter tumorresection.Wecombinedthetibialcomponentoftotalknee arthroplasty, internal fixation devices, kunstcher nail, proximal tibialockingplate,andcerclagewire,andbonecement.Thistech- niqueboneonpolyethylenesystem,enhancedwithbonecement, promises low risk of loosening.We preserved thepatellar ten- donandattacheditusingmeshtomaintainthepreviousextensor mechanismofkneejoint.Theanteriorandposteriorcruciatelig- amentandmedialcollateralligamentwerepreservedtoimprove stabilityofthekneejoint.Althoughtherewassomelittleinsta- bilityonvalgusstresstestintraoperative,butlateronthefollow upinstabilitywasgoneduetoitsfibrotictissue.Thepatientalso didnotcomplainanysymptomsandcouldwalkfullweightbear- ingwithoutanyadditionalaid.Finally,in ordertodecreasethe riskofinfection,medialgastrocnemiusrotationalflapwasused.

Thistechnique, bone onpolyethylene, wascreatedtomake an alternativetomodularendoprosthesis.Suchprocedurewouldbe functionalduetohighadaptabilityinpaediatricpatients.Further- more,itmaydecreasethenumberofsurgeriesuntiltheﬁnalgoal

(5)

Fig.6. ConstructionofTheImplant.WedrilledtheinsertcomponentofthetibialtomakeaholelaterforsuturingtheremainingACLandPCL.WeaddedK-nailbelowthe stemofbaseplatetibiaandcombineditwithproximaltibialplateusingcerclagewire.

Fig.7.PostpatellartendonandMCLreconstruction.Wesuturedtheremainingpatellartendon,MCL,ACLandPCLusingpolyestersuturetotheMesh.

(6)

CASE REPORT – OPEN ACCESS

Fig.8. Finalresultwithmedialgastrocnemiusﬂap.

Fig.9. Postoperativexray.

(7)

Fig.10. Histopathologyfeatureshowedosteosarcomapostneoadjuvantchemotherapywith75%necrosisofthetumorcellconsistedwithHUVOS2.(H&E,400×).

Fig.11.Clinicalpictureshowedalumponpreviousscar.

oflimbequalizationand adult-typetumorprosthesisimplanta- tion.

Complicationsofendoprosthesesincludeinfection,asepticloos- ening,soft tissuefailureandfracture oftheprostheses orbone.

Local recurrenceand aseptic loosening often leadto secondary amputationandrevision,whichareamajorconcernforprosthetic failure.Therefore,negativemarginsshouldbeachievedintumor resectionandchemotherapyinordertopreventlocalrecurrence.

Complicationsresultingfromlocalrecurrenceoccurredin9.3%of patients.Bothlocalrecurrenceanddeepinfectionarethetwomain causesofsecondaryamputationandrevision.Inmostcases,local

recurrenceoccurredwithinthreeyears aftersurgicalprocedure [1,5,6,9,13].

4. Conclusion

Combination chemotherapy and limb-salvage surgery in osteosarcoma gives good survival rate outcome. This bone on polyethylenehemiarthroplastysystemenablesgoodandreliable functional outcome while maintaining the knee joint for daily activity. It can be chosen as one of viable options in treating osteosarcomaaroundthekneejointforchildren,besidesdoinga

(8)

CASE REPORT – OPEN ACCESS

Fig.12.A.Tumorexposed.B.Grosspathologyofthetumor.

Fig.13.Histopathologyresultshowedthesameappearancewasobservedatrecurrencesite(H&E,100×).

Fig.14. Clinicaloutcomeafter1yearofsurgery.Patientcanwalkwithoutanyadditionalaidandnopain.Flexionandextension10–50degrees.

(9)

modularendoprosthesis.Itcostslessthanmodularendoprosthesis andcanbedoneinmanytypesofhospital.Inthatway,children patientswithosteosarcomacanbetreatedearlyandhaveabetter survivalrate.

DeclarationofCompetingInterest None.

Funding

Nonedeclared.

Ethicalapproval

EthicalapprovalhasbeenreceivedfromCiptoMangunkusumo Hospital,Jakarta,Indonesia.

Consent

Writteninformedconsentwasobtainedfromthepatient’spar- entsforpublicationofthiscasereportandaccompanyingimages.

AcopyofthewrittenconsentisavailableforreviewbytheEditor- in-Chiefofthisjournalonrequest.

Authorcontribution

YogiPrabowoperformingtheprocedure,studyconcept,data collection.

MuhammadRizqiAdhiPrimaputra:datacollection,writingthe paper.

EvelinaKodrat:datacollection.

Registrationofresearchstudies

Thisstudy hasbeenregisteredat researchregistry.com(UIN:

researchregistry5169).

Guarantor YogiPrabowo.

Provenanceandpeerreview

Editoriallyreviewed,notexternallypeer-reviewed.

Acknowledgement

Authors would like to give thanks to the Department of OrthopaedicandTraumatology,UniversityofIndonesiaandCipto MangunkusumoHospitalformakingthiscasereportdeliverable.

Nopatientorauthordetailsareincludedintheﬁgures.

References

[1]J.Y.Yang,F.W.T.Cheng,K.Wong,V.Lee,W.Leung,M.M.Shing,etal.,Initial presentationandmanagementofosteosarcoma,anditsimpactondisease outcome,HongKongMed.J.15(2009)434–439.

[2]W.Bi,W.Wang,G.Han,J.Jia,M.Xu,Osteosarcomaaroundthekneetreated withneoadjuvantchemotherapyandacustom-designedprosthesis, Orthopedics36(2013)444–450.

[3]E.S.Hart,Rightproximaltibiaosteosarcoma,Orthop.Nurs.30(2011)344–345.

[4]S.H.Chung,D.Jeon,W.H.Cho,W.S.Song,C.Kong,S.H.Cho,etal.,Temporary hemiarthroplastywithasyntheticdeviceinchildrenwithosteosarcoma aroundthekneeasabridgingprocedureuntilskeletalmaturity,J.Surg.Oncol.

112(2015)107–114.

[5]J.Huang,W.Bi,G.Han,J.Jia,M.Xu,W.Wang,Themultidisciplinarytreatment ofosteosarcomaoftheproximaltibia:aretrospectivestudy,BMC

Musculoskelet.Disord.19(2018)1–6.

[6]C.Zhang,J.Hu,K.Zhu,T.Cai,X.Ma,Survival,complicationsandfunctional outcomesofcementedmegaprosthesesforhigh-gradeosteosarcomaaround theknee,Int.Orthop.1(2018)1–12.

[7]R.A.Agha,M.R.Borrelli,R.Farwana,K.Koshy,A.J.Fowler,D.P.Orgill,etal.,The SCARE2018statement:UpdatingconsensusSurgicalCAseREport(SCARE) guidelines,Int.J.Surg.60(2018)132–136.

[8]L.Aponte-tinao,M.A.Ayerza,D.L.Muscolo,G.L.Farfalli,Survival,recurrence, andfunctionafterepiphysealpreservationandallograftreconstructionin osteosarcomaoftheknee,Clin.Orthop.Relat.Res.1(2014)1–8.

[9]P.X.Tan,B.C.Yong,J.Wang,G.Huang,J.Q.Yin,C.Y.Zou,etal.,Analysisofthe efﬁcacyandprognosisoflimb-salvagesurgeryforosteosarcomaaroundthe knee,Eur.J.Surg.Oncol.38(2012)1171–1177.

[10]M.E.Puhaindran,R.W.H.Pho,Biologicalreconstructionforchildrenwith osteosarcomaaroundtheknee,Ann.Acad.Med.Singapore43(2014) 499–505.

[11]S.Kang,J.S.Lee,J.Park,S.Park,Stagedlengtheningandreconstructionfor childrenwithaleg-lengthdiscrepancyafterexcisionofanosteosarcoma aroundtheknee,BoneJointJ.99(2017)401–408.

[12]X.He,H.Zhang,Y.Hu,Limbsalvagebydistractionosteogenesisfordistal tibialosteosarcomainayoungchild,Orthop.Surg.8(2016)253–256.

[13]N.W.Lang,G.M.Hobusch,P.T.Funovics,R.Windhager,Whatsportsactivity levelsareachievedinpatientswithmodulartumorendoprosthesesof osteosarcomaabouttheknee?Clin.Orthop.Relat.Res.473(2015)847–854.

[14]M.M.Malawer,P.H.Sugarbaker,MusculoskeletalCancerSurgeryTreatmentof SarcomasandAlliedDiseases,LippincottWilliams&Wilkins,USA,2001,pp.

483–502.

[15]M.M.Malawer,J.C.Wittig,B.Jacob,OperativeTechniquesinOrthopaedic SurgicalOncology,2ndEditio,LippincottWilliams&Wilkins,USA,2015.

[16]F.H.Sim,P.F.Choong,K.L.Weber,MasterTechniquesinOrthopaedicSurgery:

OrthopaedicOncologyandComplexReconstruction,1stEditio,Lippincott Williams&Wilkins,USA,2011.

OpenAccess

ThisarticleispublishedOpenAccessatsciencedirect.com.ItisdistributedundertheIJSCRSupplementaltermsandconditions,which permitsunrestrictednoncommercialuse,distribution,andreproductioninanymedium,providedtheoriginalauthorsandsourceare credited.