Rev. bras. ortop. vol.52 número5

r e v b r a s o r t o p . 2017;52(5):625–627

SOCIEDADE BRASILEIRA DE ORTOPEDIA E TRAUMATOLOGIA

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Case

Report

Chronic

recurrent

multifocal

osteomyelitis:

a

case

report

夽

Adayellen

Cristina

Salomé

Oliveira

_,

_Andrew

_Raimundo

_Fernandes

_da

_Costa

_,

Ana

Maria

Magalhães

Valle

Cundari

_,

_Vinícius

_Bicalho

_Rodrigues

a_{UniversidadedeItaúna,Itaúna,MG,Brazil}

b_{CasadeCaridadeManoelGonc¸alvesdeSousaMoreira,Itaúna,MG,Brazil}

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Articlehistory:

Received5July2016 Accepted25August2016

Availableonline4September2017

Keywords:

Osteomyelitis/pathology Osteomyelitis/diagnosis Diagnosticimaging

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Chronicrecurrentmultifocalosteomyelitis(CRMO)isarareidiopathicinflammatory dis-easethataffectsmainlychildrenandyoungadults.Theclinicalsignsandsymptomsare nonspecific,hinderinganddelayingdiagnosis.Radiologicalandhistopathologicaltestsare essentialforitsdefinition.AcaseofCRMOisreported,demonstratingtheimportanceof clinical,laboratory,andradiologicaldatafordiagnosis.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublishedbyElsevierEditora Ltda.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http:// creativecommons.org/licenses/by-nc-nd/4.0/).

Osteomielite

crônica

multifocal

recorrente:

relato

de

caso

Palavras-chave:

Osteomielite/patologia Osteomielite/diagnóstico Diagnósticoporimagem

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Osteomielitecrônicamultifocalrecorrente(OCMR)éumadoenc¸ainflamatóriaidiopática raraqueacometeprincipalmentecrianc¸aseadultosjovens.Ossintomasesinaisclínicos sãoinespecíficos,dificultameretardamodiagnóstico.Osexamesradiológicose histopa-tológicossãoindispensáveisparasuadefinic¸ão.Nesterelato,osautoresapresentamum casodeOCMRquedemonstraaimportânciadosdadosclínicos,laboratoriaiseradiológicos paraodiagnóstico.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublicadoporElsevierEditora Ltda.Este ´eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND(http:// creativecommons.org/licenses/by-nc-nd/4.0/).

夽

PaperdevelopedatUniversidadedeItaúna,Itaúna,MG,Brazil.

∗ _{Correspondingauthor.}

E-mail:andrewcosta.md@gmail.com(A.R.Costa).

http://dx.doi.org/10.1016/j.rboe.2017.08.017

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Introduction

Chronicrecurrentmultifocalosteomyelitis (CRMO)isarare idiopathic inflammatorydisease,the diagnosis ofwhich is made by exclusion, affecting mainly children and young adults.Theearlyidentificationofthispathologyavoids unnec-essary examinationsand prolonged antibiotictherapy. The clinicalsignsandsymptomsofthediseasearenonspecificand hinderthediagnosisbasedonclinicalpresentationonly.Thus, radiological and histopathologicalexams are indispensable foritsdefinition.

Theobjectiveofthisreportis,therefore,topresentacase ofCRMO,and todemonstratethe diagnosticimportanceof clinical,laboratoryand,mainly,imagingtests.

Case

report

A 44-year-old female patient, who suffered a car accident 12 years before, with no fractures; but had a laceration on the right thigh. After four years, she presented with continuouspainintheleftleg,withnocturnalworsening. Pre-viouslyhealthy,shedeniedafamilyhistoryofmusculoskeletal

conditions, drug reactions, previous surgical history and febrileconditionsaftertheaccident.

OnSeptember2005,shesoughtmedicalcareand under-went computed tomography (CT) (Fig. 1), which showed corticalthickeninginthelefttibialshaftwithareasofbone resorption. She underwent surgical treatment, progressing withtibialfracture threedaysafterthe procedure,the rea-sonofwhich,accordingtothepatient,wastheremovalofa largebonefragmentduringthesurgicalprocedure.Treatment followed withcastimmobilizationanduseofnon-steroidal anti-inflammatorydrugs(NSAIDs).

OnOctober2013,thepatientsoughtmedicalcarebecause had pain inthe right leg forthree months, similar to the previous onein theleft leg. Imagingand histopathological examswereperformed.Radiographsoftherightlegshowed diffuse corticaland medullary sclerosis at the tibial shaft. ThesechangeswereconfirmedinaCT,whichalsorevealed aminimalareaofboneneoformationwithcortical thicken-ingattheproximalendofthefibula.Surgicalbiopsyshowed anonsepticlesion,andthemedullarycanalwasrecanalized. MDP-Tc99m _{bonescintigraphydemonstratedhypercaptation}

oftheradiopharmaceuticalagentinthemiddlethirdofthe rightleg,withoutdenotingboneinvolvementduetoan infec-tiousprocessorindicatinganyotherasymptomaticfocus.

Fig.1–Axialcomputedtomographyimagesofthemiddlethirdofthelegbonesshaftshowperiostealandendostealbone

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TheanalysisoftheexamsallowedthediagnosisofCRMO. Thus,thepatientreceivedNSAIDtherapy,andhasnot pre-sentedanyrecurrenceofpain.

Discussion

CRMO is an idiopathic inflammatory bone disorder seen mainlyinchildrenandadolescents1_{;however,ithasalready}

beendescribedinolderpatientsupto55years.2,3_Thisisarare

variantofosteomyelitis,correspondingto2–5%ofthecases.4

Patientsusuallypresentwithinsidiousonsetofheat,pain,and soft-tissueedema,restrictedtooneormorebones.5_Thelower

limbsarethemostaffected,5_{withmetaphysesorequivalent}

sitesbeingthemostcommonlocations.6

Itsdiagnosisismadebyexclusion,and themaincauses tobeexcludedareneoplasmsandinfections.7,8_Itisbasedon

thefollowingcriteria:(a)bonelesionswithimagessuggesting subacuteorchronicosteomyelitis;(b)unusuallocationwhen comparedtoinfectious,multifocalosteomyelitis;(c)absence ofabscess,fistulaorsequestra;(d)absenceofanetiological agent;(e)nonspecifichistopathologicaland laboratorytests consistentwithsubacuteorchronicosteomyelitis;(f)typical episodesofprolongedandrecurrentepisodesofpain,and(g) occasionalskinmanifestations,suchaspalmoplantar pustu-losis,acne,psoriasisvulgarisandpyodermagangrenosum.9,10

Thepatientpresentedwithalltheabovecriteria,exceptfor cutaneousmanifestations.

Image exams reveal nonspecificalterations that require histopathologicalexams tobetterdefinethe disease.These testsruleoutthepossibilityofneoplasticorinfectiouslesions, whiletheimagesareusefulforassessingtheextentofthe dis-ease,reveallyticareasandperiostealreaction,whichevolveto hyperostosisandsclerosis,exactlywhatthepatient’simages showed(Fig.1).

Treatment involves specially NSAIDsthat aimat symp-tomaticrelief.Antibioticsareconsideredineffective.Thecase patientwastreatedwithNSAIDsandhadgoodprogress,with norecurrenceofpain.

CRMOprogressionisunpredictable.Althoughmostcases resolve spontaneously in months to years, there are

reports of symptomatic patients for up to 25 years after diagnosis.

KnowledgeoftheappearanceofCRMOlesionsandtheir typicalchangesisimportantforanearlydiagnosis,leadingto areductioninthenumberandnecessityofbiopsies,surgeries andantibiotictherapy.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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