Spontaneous healing of a traumatic critical radius ... - iKnow

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International Journal of Surgery Case Reports

j o ur na l h o m e p a g e :w w w . c a s e r e p o r t s . c o m

Spontaneous healing of a traumatic critical radius bone defect in adolescent: A rare case report

Aryadi Kurniawan

^∗,1

, Triadi Wijaya, Witantra Dhamar Hutami

DepartmentofOrthopaedicsandTraumatology,CiptoMangunkusumoNationalCentralHospitalandFacultyofMedicine,UniversitasIndonesia,Jalan DiponegoroNo.71,JakartaPusat,Jakarta10430,Indonesia

a rt i c l e i nf o

Articlehistory:

Received24February2021

Receivedinrevisedform18March2021 Accepted18March2021

Availableonline22March2021

Keywords:

Criticalbonedefect Spontaneoushealing Adolescentbonedefect Rarecase

Casereport

a b s t ra c t

INTRODUCTIONANDIMPORTANCE:Fracturewithacriticalbonelossisassociatedwithaprofoundburden ofdiseaseimpact.Althoughthereareseveraloptionsexistforitstreatment,butstillthosereconstructive proceduresaretechnicallydemanding,relativelyexpensiveandsometimestheresultislessthanwhat wasexpected.Theobjectiveofthisstudyistoreportararecaseofspontaneoushealingofacriticalradial bonedefectinanadolescent.

CASEPRESENTATION:Wereporteda15yearoldboywithasegmentalopenfractureofleftradius,open fractureofleftdistalshaftulnaandclosedfractureofleftintercondylarhumerus.Themiddlefragment ofafracturedradiuswasextrudedout,pulledoutandthenthrownawaybyhisparent.Debridement, openreduction,andinternalfixationforulnawereperformedaswellasrepositionandinternalfixation fortheintercondylarhumerusfracture.Theplanwastowaituntiltheulnarfractureandintercondylar fracturetohealwithoutanysignofinfectionandproceedtoovercometheradialcriticalbonedefect.

ThiscasereporthadbeenreportedinlinewithSCAREcriteria.Thepatientshowedupsevenmonthslater withsolidunionofthecriticalradiusbonedefectandfullyfunctioninghandwithonlyslightlimitation inpronation.

CLINICALDISCUSSION:Osteogenesisinfracturerequiresosteogeniccells,osteoinductivecomponents, osteoconductivescaffold,andstability.Despitethefactthatcriticalbonedefectposesgreatchallenge foritsmanagement,intactperiosteumandsufficientsofttissueperfusionwereabletoprovidethose biologicrequirementsadequatelyforfracturehealingandensurespontaneoushealingofatraumatic criticalbonelossinadolescentwithoutanyreconstructiveprocedure.

CONCLUSION:Spontaneoushealingincriticalbonedefectispossible,providedallthefavorablefactors presenttosupportthisphenomenon.

©2021TheAuthors.PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.Thisisanopen accessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

1. Introduction

Fracturesassociated withbone lossisasignificantchallenge whichfrequentlyrequirerepeatedsurgicalinterventions.Despite thosenumerousattemptstoovercomeboneloss,theresultswere frequently less than what we hope for. Traumatic bone lossis definedastheexpulsionordissapearanceofbonefragmentdue totraumaorremovalofdevitalizedboneduringdebridement[1].

Generalagreementforthedefinitionofcriticaltraumaticboneloss is whenthesizeof thedefect is2–3timesthediameterofthe involvedbone[2].Maufreyetal.[3]statedthatacriticalbonedefect generallyhascircumferentialloss>50%oralossinlengthof>2cm.

Whilefracturehasitspotencyforselfhealing,especiallyinchil- drenandadolescent,theexistenceofcriticalbonedefectmaylead tononunionduetolimitationofmusculoskeletalsystemabilityto

∗Correspondingauthor.

E-mailaddress:aryadi.kurniawan@ui.ac.id(A.Kurniawan).

1 Firstauthor.

fillthedefectsandrepairthefractureunlessreconstructivebony surgeryisperformed.

Historically,afterseveralsurgicalattempswereperformedand theresultwasnotfunctionallimbthentheextremitywasconsid- eredmangledandsubsequentlymanagedbyamputationwitha consequenceofaconsiderablelossofqualityoflife.Nowadays,the epicentrumofmanagementhasshiftedtowardlimbsalvagepro- cedureswhichencompassesfollowingoptions:boneshortening, distractionosteogenesis,theuseofvascularizedandnonvascular- izedbonegraftsandinducedmembrantechniques[3].

Giannoudisetal.[4–6]statedtheDiamondConceptasthebasic requirementforfracturehealingwhichconsistofosteogeniccells, osteoinductive,osteoconductiveandmechanicalstability.Thebio- logical componentof suchdiamond conceptwere deliveredby an intact and adequate soft tissue coverage. Giannoudis et al.

[4,6]alsostatedthataccelerationoffracturehealingorresolving delayedunionandnonunionispossiblebutithastopassthrough adequacyofsuchbiologicalcomponentofdiamondconceptand supportedwithsufficientmechanicalstability.Regardlesswhich surgicaltechniquesbeingused,thosereconstructivesurgeriesto

https://doi.org/10.1016/j.ijscr.2021.105806

2210-2612/©2021TheAuthors.PublishedbyElsevierLtdonbehalfofIJSPublishingGroupLtd.ThisisanopenaccessarticleundertheCCBYlicense(http://creativecommons.

org/licenses/by/4.0/).

Fig.1.ClinicalFindingduringHospitalAdmission.Therewereswellingandangu- lationoftheleftdistalforearm,andanopenwound.

overcomecriticalbonelosswereperformedinanefforttodeliver biologicalsubstanceandmechanicalfixationrequiredforosteoge- nesis.

Thiscasereportdescribesararecaseofaspontaneoushealingof acriticalradialbonedefectinanadolescentduetotrauma.Theaim ofthisstudyistoconfirmthatsuchspontaneoushealingincritical bonedefectispossible,providedtheperiosteumisintact,thesoft tissue coverageisadequateandthereisnoinfection.Therefore, itisvitaltofulfillthoserequirementsinmanagementofcritical bonedefect.ThiscasereporthadbeenreportedinlinewithSCARE criteria[7].

2. Patientinformation

Wereporteda15yearoldboywithhistoryoffallingfromatree 26hpriortoadmission.Hefellwithhisleftelbowinfullextension andhadanopensegmentalfractureofleftradius,openfracture of leftdistal shaftulnaand closedfracture ofleftintercondylar humerus.Themiddlefragmentofradiusfracturewasextrudedout- wardandwasthenpulledoutandthrownawayunpurposelybyhis parentbecausemistakenlyidentifiedthebonefragmentaswood thatpiercingtohisson’sforearm.Atthetimehecametohospital, hedidn’tbringtheremainingbonefragment.

3. Clinicalfindings

Physicalexaminationatthetimeofadmissionrevealedswelling andangulationontheleftdistalforearmwithanopenwoundsize 3×0.5cm.Therewasalsoaswellingattheelbowjointwithno openwound.Distalperfusionandsensorywasnormal(Fig.1).

4. Timeline

Time ClinicalFinding Treatment

Twentysixhours beforehospital admission

Openfracturewithbone fragmentextrudedfromthe skinandwaspulledout

Woundtoilet,primary sutureofthewound, antibiotic

Twentysixhours aftertrauma

Pain,swelling,angulationand openwoundattheleftdistal forearmwithpreserved perfusionandsensory.Motoric waslimitedduetopain Swellingandpinattheleft elbow

Emergency

debridementandback slabapplication

Sevendaysafter admission

Pain,swelling,andangulation attheleftdistalforearmwith preservedperfusionand sensory.Motoricwaslimited duetopain.Swellingandpinat theleftelbow.Woundwasleft openduetosofttissueswelling

Openreduction internalfixationof ulnarfractureand intercondylarfracture, applicationofbackslab

Fig.2. BoneDefectMeasurement.Grossanatomically,thebonelosswas9cm length.Thepercentageofbonelossaccordingtothemeasurementis38%,calculated fromthe21boxeslossofbonecomparedto54boxeslengthofradius.

5. Diagnosticassessment

Anteroposteriorand lateralviewradiographsof leftforearm showedtransversefractureofdistalshaftulnawithdisplacement toside (Fig. 2). Therewas 9 cm (38%) bone loss ofradius that startedfrommidshaftradiustometaphysisofdistalradius.The elbow’santeroposteriorandlateralviewradiographsshowedthat thispatientalsogotintercondylerhumeralfracturewithT-shaped fracturelinethatextendingtometaphysisofdistalhumerusbut therewasnosignificantdisplacementofthefragments.

Wediagnosedthepatientwithopensegmentalfractureofleft radiuswithboneloss,openfractureofleftdistalshaftulnaGustillo- andersongradeII,andclosedfractureofleftintercondylarhumerus RadinsRiseboroughtypeII.

6. Therapeuticintervention

Two-stage surgeries were carried out, with the emergency debridement and application of backslab performed at emer- gencyoperatingtheatreandthewoundwasleftopenduetosoft tissueswelling.Secondsurgerywasperformedaweekafterdur- ingwhich openreductionandinternalfixationofulnarfracture andintercondylarhumerusfracturewereperformed,stillwiththe applicationofbackslabpostoperatively.Theradiusbonedefectwas leftuntouchedtoconfirmtherewasnosubsequentsignofinfection

Fig.3.SevenMonthsPostoperativeClinicalCondition.Onlylimitedpronantionwasfound.

Fig.4. SequentialXRayFindingsofthePatient.(Left)atinitialencounter,theradialbonedefectwasmeasuredas9cm(38%)boneloss.(Middle)ataweekafterinitial encounter,theulnarfracturewasfixatedusingplateandscrew.(Right)sevenmonthsafterinitialencounter,thebonedefecthadbeenfilledwithnewboneformation.

andwasplannedtohavereconstructivesurgeryaftertheulnarand intercondylarfracturehealed.

7. Followupandoutcomes

Patientattendedoutpatientclinicforwoundcareonceaweek severaltimes,onlytostopcomingafterthewoundhadhealedand cameagain7monthsafterwards.At7monthsfollowup,patient

hadnopainnordeformityontheforearmandelbow.Therange ofmotionofthewristandelbowwassummarisedinTable1and shownatFig.3.

Patienthad normalwristflexion,extension,radialdeviation, ulnar deviation, and supination but limited pronation. The 7- months postoperative x ray showed the fracture of ulna and intercondylarhumerushadbeenunitedandtheradiuscriticalbone defectwasfilledwithsolidbonewithnogrossangulation(Fig.4).

Table1

MeasurementofWristRangeofMotionSevenMonthsafterInjury.

Variables Values NormalValues

WristProfiles

Radialheight 8mm 8–17mm

Ulnarvariance +1cm −4to+2mm

RadialInclination 18^◦ 16^◦–29^◦

Palmartilt 28^◦(dorsal) 0^◦–22^◦(palmar)

RangeofMotionofWristJoint

Flexion 0^◦–80^◦ 0^◦–80^◦

Extension 0^◦–70^◦ 0^◦–70^◦

Pronation 0^◦–40^◦ 0^◦–90^◦

Supination 0^◦–90^◦ 0^◦–90^◦

Ulnardeviation 0^◦–30^◦ 0^◦–30^◦

Radialdeviation 0^◦–20^◦ 0^◦–20^◦

8. Discussion

Mostpatientswithtraumaticbonelosswillcometoemergency roomasanemergencyopenfracturecase[8].Itisimportantto carefullyassesstheperfusionanddegreeofcontaminationofthe woundsothatadequateandpropermanagementcanbedelivered.

Thegoalsoftreatmentarenotonlytoachievebonyunionbutalso restoration offunctionassoonaspossible.Duetodevelopment of bonedefectmanagement methodand softtissue reconstruc- tiontechnique,criticalopenbonelosswasmostlytreatedwithtwo stagetreatment.Firststageismeticulousdebridementandremoval ofdevitalisedtissuefollowedbyreconstructionofsofttissue,ifnec- essary,toensuresufficientcoverage.Debridementitselfmaycreate abonedefectorevenextendtheexistingboneandsofttissuedefect.

Oncethesofttissuecoverageandinfectionissueshavebeenfully addressed,thesecondstageisbonereconstructionsurgerytofillin thebonegapwhichmaybeoneoftheseoption:boneshortening, distractionosteogenesis,theuseofvascularizedandnonvascular- izedbonegraftsandinducedmembrantechniques[1,3].Regardless thetechniquebeingused,treatmentofcriticalbonelosshastoful- fillpreservationoflimblengthandalignment,solidunionofbone andrestorationoffunction.

Autogenousbonegraftasthegoldenstandardmanagementfor bonegaphasbeensuccessfullytreatedbonedefectlessthan5cm.It providesgrowthfactors,osteogeniccellsandallowsforearlyrevas- cularizationthateventuallyleadstoahighincorporationrate.The challengeforautogenousbonegraftisthelimitedavailability,espe- ciallyinchildren,anddonorsitemorbidity.Thechallengegrows biggerwhenwearedealingwithalargebonedefect.Vascularized bonegraftcantreatadefectuntil10–20cmlength[3]butitrequires skilldemandingmicrosurgery,longtermimmobilization,andreha- bilitation.Otherdisadvantagesofthismicrosurgeryarepossibility ofnon-unioninthedockingsite,stressfracturesofthegraftanda lengthyperiodforgrafttogrowandreachthedesireddimension.

Distractionosteogenesisisanotheroptionoftreatmentforbone lossandmaysucesfullymanageuntil10cmofbonedefect[3].The majordrawbacksarethistechniqueiscumbersomeforthepatients, possibility ofrecurrentpintractinfection,requirementof addi- tionalsurgeriesforfixatorre-alignmentsandwirere-tensioning.

Additionalsurgeriesarealsofrequentlyneededfordebridement andpromoteunionatthedockingsite.Fromthepatientsside,this treatmentrequiresverygoodpatientcomplianceduetoitslong andfrequentsurgicalintervention[1,9].

One major difference between adult and pediatric in bone regeneration is the characteristic of periosteum. Periosteum in pediatrichashugeosteogeniccapacitywhichisgeneratedbyosteo- progenitorcellsinitsinnerlayer,aswellastheabilitytodeliver osteoinductivesubstances.Thepresenceofperiostealsleevewhich bridgethefracturegapisimportantinspontaneoushealingofbone loss.Inadults,periosteumismuchweaker,thinnerandmoreadher- enttothebonewhilein childrentheperiosteumisthickerand

lessadherentthatit willeasilybestrippedofffromtheunder- lyingbone.Thelessadherentperiosteumisoneofreasonitmay stayrelativelyintactinabonedefect[10].Inthetraumaticbone losswithrelativelyintact periosteumsleeve,thedefect willbe filledbyhematomaandosteogenesiswillbeinitiatedbytheinner layerofperiosteumthatinitiallyproducethecartilaginoustissue whichlateronwillossify[10].Thepresenceofmuscularsofttissue coverageisalsoutmostimportantsinceitensuresperfusionand deliveriesofgrowthfactors.Theabsenceofinfectionisalsopivotal becauseinfectionmayretardtheprocessoffracturehealingand sincealmostallfractureswithbonedefectstartedasanopenfrac- turetheninitialmanagementofopenfractureisveryimportant aswell.Therefore,conservativetreatmentforbonedefectisactu- allypossibleprovidedallrequiredcomponentsforfracturehealing issufficientlyavailable.Thebigquestioniswhatisthemaximum lengthforbonedefectwhichcanbemanagedconservatively?

Inourcase,patientfellfromatreeandgotanopensegmental fractureofradiuswithabonefragmentextrudingfrominsideout.

Thebonelossfrominthiscasewasduetohisparentpullingthe fragmentoutandnotfromtheinitialtrauma.Therewasnomajor softtissuecompromise andbythemechanismofboneloss,we mightassumethattheperiostealsleevewasstillrelativelyintact andthesurroundingmuscleenvelopewasstillrelativelyundam- aged.Debridementandotherpropermanagementforopenfracture wasperformedtoensurecontaminationwasfullyaddressedand infectiondidnot happen.The patientis 15 years ofage which canstillbeconsideredaswithingrowthspurtofpubertyduring whichtheosteogenesisis enhanced duetothepresence ofsex hormone.Thepresenceofsexhormoneduringgrowthspurtmay enhancefracturehealing[11]Adequatestabilitywasprovidedby openreductionandinternalfixationofulnasupportedwithback slabfor the firstfew weeks. Early mobilizationwas alsomade possiblebyopenreductionandinternalfixationoftheelbowinter- condylarfracture.Thispatienthadallofthefavorablefactorsthat supportspontaneoushealingofthecriticalradiusbonedefectand suchspontaneoushealingmayovercome9cmbonedefect(38%) ina15yearsoldboy.

9. Patientperspective

Patientandfamilyhadbeeninformedregardingthecondition, treatment,resultofthetreatment,andprognosis.Patientandfam- ilyunderstood.

DeclarationofCompetingInterest

The authors certify that They have NO affiliations with or involvementinanyorganizationorentitywithanyfinancialinter- estor non-financial interest in the subject matteror materials discussedinthismanuscript.

Sourcesoffunding

The authors received no financial support for the research, authorship,and/orpublicationofthisarticle.

Ethicalapproval

Theethicalapprovalwasnotrequiredforthiscasereport.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonrequest.

Authorcontribution

AryadiKurniawan:studyconcept,datacollection,datainterpre- tation,andwritingthepaper

TriadiWijaya:datacollection,datainterpretationandwriting thepaper

WitantraDhamarHutami:datacollection,datainterpretation andwritingthepaper

Registrationofresearchstudies

Thiscasereportisnotafirstinmanstudy.

Guarantor

AryadiKurniawan.

Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed.

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