Rev. bras. ortop. vol.52 número5

SOCIEDADE BRASILEIRA DE ORTOPEDIA E TRAUMATOLOGIA

w w w . r b o . o r g . b r

Case

Report

Total

knee

replacement

in

patients

with

diffuse

villonodular

synovitis

夽

Lucio

Flávio

Biondi

Pinheiro

Junior

,

Marcos

Henrique

Frauendorf

Cenni

,

Rafael

Henriques

Soares

Leal

_,

_Luiz

_Eduardo

_Moreira

_Teixeira

a_{RedeMaterDeideSaúde,BeloHorizonte,MG,Brazil}

b_{HospitaldaUnimed,BeloHorizonte,MG,Brazil}

c_{UniversidadeFederaldeMinasGerais,HospitaldasClínicas,BeloHorizonte,MG,Brazil}

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Articlehistory: Received8July2016 Accepted16August2016 Availableonline14August2017

Keywords:

Villonodularpigmentedsynovitis Arthroscopy

Knee

Giantcelltumors

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b

s

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Thispaperreportsacaseofdiffusepigmentedvillonodularsynovitis(DPVNS),associated withadvancedgonarthrosis,submittedtototalkneereplacement.Thepatienthad progres-sivepainandswelling.Shehadtwoprevioussurgeries,firstlyarthroscopicsynovectomyand subsequentlyopensynovectomyassociatedwithradiotherapy,withrecurrenceofthe dis-ease.Magneticresonanceimagingrevealeddiffusesynovitis,advancedarthrosis,andbone cysts.Thepatientwassubmittedtoatotalkneereplacementandsynovectomy.Therewas agoodpostoperativeclinicalcourse,withimprovementofpain,function,andjointedema onexamination.Thepatientwillbefollowedregardingthepossibilityofdiseaserecurrence andimplantsurvival.

©2017PublishedbyElsevierEditoraLtda.onbehalfofSociedadeBrasileiradeOrtopedia eTraumatologia.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http:// creativecommons.org/licenses/by-nc-nd/4.0/).

Artroplastia

total

do

joelho

em

paciente

com

sinovite

vilonodular

pigmentada

forma

difusa

Palavras-chave:

Sinovitepigmentadavilonodular Artroscopia

Joelho

Tumoresdecélulasgigantes

r

e

s

u

m

o

Estetrabalhorelataumcasodesinovitevilonodularpigmentadaformadifusa(SVNPD), associadaa genoartrose avanc¸ada, que foi submetida a artroplastiatotal dojoelho. A pacienteapresentavadoreedemaemjoelhodecaráterprogressivo,jásubmetida previ-amenteaduassinovectomias,umaporviaartroscópicaeoutraporviaaberta,alémde radioterapia,comrecidivadadoenc¸a.Asradiografiasdemonstravamobliterac¸ãodosespac¸os

夽

PaperdevelopedatthenetworkMaterDeideSaúde,BeloHorizonte,MG,Brazil. ∗ _{Correspondingauthor.}

E-mail:[email protected](L.F.PinheiroJunior).

http://dx.doi.org/10.1016/j.rboe.2017.08.002

articulares,alémdeerosõesecistosintraósseosnatíbiaenofêmur.Ressonânciamagnética evidenciousinovitedifusaextensa,alémdeartroseavanc¸adaecistosósseos.Apacientefoi submetidaaartroplastiatotaldojoelhocombinadacomsinovectomiaampla.Ela apresen-touboaevoluc¸ãoclinicapós-operatória,commelhoriadador,dafunc¸ãoedoedema.A pacienteseráacompanhadaquantoàpossibilidadederecorrênciadadoenc¸aesobrevida doimplante.

©2017PublicadoporElsevierEditoraLtda.emnomedeSociedadeBrasileirade OrtopediaeTraumatologia.Este ´eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND (http://creativecommons.org/licenses/by-nc-nd/4.0/).

Introduction

Pigmentedvillynodularsynovitisisarare, benignand pro-liferativediseaseofthesynovialtissueofuncertainetiology thatcandeterminethedestructionofjointcartilageandresult inosteoarthrosis.1_{Simondescribedthelocalizedtypeofthe} knee andMoser, in1909,described the diffusetypeofthe disease.Jaffeetal.2_{proposedthetermpigmented} villonodu-larsynovitisforthesemanifestations,butthenomenclature proposedbyGranowitzdefinedthatthetermpigmented vil-lonodularsynovitisshouldbeusedforintra-articularlesions, pigmented villonodular bursitis for lesions located in the bursaeandpigmentedvillonodulartenosynovitisforlesions originatingfromtendinoussheaths.

Thispathologycanbedividedintotwotypes:localizedand diffuse.Themostcommontypeisthediffuseone.Itismore frequentbetween20and50yearsofagebutcanreachanyage, withaslightprevalenceinfemales.Thetreatmentaimsatthe resectionofthelesion,botharthroscopicallyand/orviaopen surgery,withlocalrecurrencebetween10%and56%.3

Fig.1–Clinicalaspect(A),magneticresonanceimaging(B)andradiographs(C),whichdemonstratediffusesynovitis, arthrosis,andbonecysts.

This work describes a case of diffuse villonodular pig-mentedsynovitisaffectingtherightknee,thatwasassociated with advanced gonarthrosis and underwent total replace-ment.

Clinical

case

Fig.2–Imageswhichdemonstratepreoperativeaspect–DPVNS,boneflawsandbonesectionsbefore(A)andafter(B) graftingandwidesynovectomy.

110◦_{.Thekneeradiographshowedadvanced}

tricompartmen-talarthrosis,grade4ofKellgrenandLawrence,asignificant lossof joint space and the presenceofvarious epiphyseal bonecystsinthetibiaandfemur.Magneticresonanceimaging demonstratedadvancedtricompartmentalarthrosis,aswell asexuberantdiffusesynovitisthroughoutthejoint, includ-ingpoplitealfossaandextra-articularextensionsintendinous sheaths,inadditiontovariousbonecystsinthefemurand tibia(Fig.1).

The patient underwent total joint replacement of the knee,associatedwithawideperioperativesynovectomy.Bone defects resulting from the cysts, which formed contained defects, were found; bone grafts were put in the defects (Fig.2). Theposteriorcruciateligament wassacrificedwith aposteriorsacrificefemoralcomponent(PS),and,inthiscase, non-replacementof the patella was chosen. We also used intravenous tranexamic acid in anesthetic induction and 15min before releasing the tourniquet due to the risk of increasedbleedingfrom surgery. Thepatientleft the oper-atingroomingoodconditions,andremainedintheICUon thedayofsurgery,beingtransferredtotheroomonthefirst postoperativeday(POD).Shewasdischargedonthesecond POD.

Non-weightbearingwasmaintainedfor30daysduetobone graftingonthemedialfemoralcondyle.Thepatienthadagood postoperativeevolution,despitetheshortfollow-up.Shehas goodlimbalignment,fullextensionand100◦_{flexion.Shealso}

reportsimprovementofpainandregressionofkneeedema (Figs.3and4).

Discussion

Pigmentedvillonodular synovitisisarare disease,with an incidenceof1.8casespermillion,andshouldbesonamed whenfound inintra-articularlocations.This lesioncan be foundinoneineach2500arthroscopies.4_{Themostcommon} locationisintheknee,about80%,followedbytheankle. Intra-articulardiseasemaybelocalizedordiffuse,withthefirsttype beingalmostexclusivelyfoundintheknee.DPVNSis char-acterizedbysynovialortendinoussheathhyperplasia,with

markedproliferationofstromalcells,largeamountsofintra andextracellularhemosiderinandmultinucleatedgiantcells. Thediseaseismoreprevalentinpatientsbetweentheagesof 20and50,butitcanaffectanyage.Thelocalizedtype repre-sents6%ofthetotalofthisdiseaseandhasamildpreference forfemales.5_{AccordingtoDinesetal.,}6_Hoffafat, suprapatel-larrecess,intercondyleand posteriorcapsuleare themost commonlocations,thelattertherarer.

DPVNShasunknownetiology,althoughtrauma, inflamma-toryprocesses,neoplasmsandlipidmetabolicdiseaseshave beenimplicatedasthecauseofthedisease.Recent cytoge-neticstudiessuggestevidencemorerelatedtoneoplasia.3,7

In diffusetypes, repetitivejoint effusion, limited range of motion and pain are common symptoms. In the local-izedtypesthemechanicalsymptomsthatsimulatemeniscal lesionsandinstability,palpablemassesandpainarefrequent complaints andoften makediagnosis difficult. Inposterior lesions,painisoftenreportedwithkneeflexion.8_{Pain and} tumorareusuallyprogressive.Thetimefromonsetof symp-tomstodiagnosisisonaverage19monthsinthelocalized typesand15monthsinthediffuseones.Thecorrelationof symptomonsetandtraumaispresentin44–53%ofpatients.5 Complementarytestsaidinthediagnosis,andmagnetic resonanceimagingisthemostsensitive.3,4,7_{Radiographsare}

Fig.4–ClinicalaspectandROMofthepatientwithonemonthaftersurgery.

usuallynormal,butdegenerationofthejointsurfacemaybe presentandismorefrequentinthehipandshoulderjoints. Intheknee,thesechangesarerare,butthenarrowingofjoint spaceandosteophytesarefindingsrelatedtoDPVNS,asfound inthiscase.Recentstudiessuggestthatproteolyticenzymes producedbygiantcellswithinthehyperplasticsynovialtissue couldplay arole inthisjoint degradation.4 _Magnetic reso-nanceimagingisanimportanttesttoestablishthediagnosis anddirecttreatment.AreasoflowsignalonT1andT2are observedinthesynovialmembrane,whichisirregular,and theassociationwithjointeffusionisfrequent.7

Thehistologicalfindingsareofawell-differentiatedlesion, withdestructive proliferation ofsynovial-like mononuclear cells,associatedwithmultinucleatedgiantcells, xanthoma-tous macrophages, presence of hemosiderin, lipids and lymphoplasmacytic inflammatorycells.In localizedlesions thereisthepresenceofapseudocapsule.4,7

DPVNS is a progressive benign lesion, and malignancy israre.1 _{The recommended treatment forDPVNS islesion} resection.Itcan bearthroscopicalor open.In diffusetype, arthroscopyhasagoodindicationwiththe useofmultiple portals.Theuseofposteriorarthrotomymaybenecessaryin casesofposteriorfossadisease.Inthelocalizedtype,the par-tialarthroscopicsynovectomyinwhichthelesionisremoved isless invasive,but intumorswell-delimited bya pseudo-capsule,open resectionisa goodoption.6,7 _{Relapsesrange} from10%to56%,andaremorecommonindiffusetypeand maytakeyearstooccur.5_{Therecurrenceinlocalizeddisease} isaround3%.9_{Radiotherapy isusedincaseswithmultiple} recurrences.Malignancyoccursin3%ofcasesandcorrelates withmultiplerecurrencesandradiotherapy.Patientfollow-up isdonewithperiodicmagneticresonanceimagingeverysixto 12months.7

Hamlinetal.10 _{followed 18patients atMayo Clinicwho} were diagnosed with DPVNS, and underwent total knee arthroplasty,14withthediffusetype–activein11andinactive inthree–andfourwiththelocalizedtype.Themean follow-upwas9.9years.Allpatientswiththediffuseactivetypealso underwenttotalsynovectomy.Attheendofthefollow-up,in 14ofthe18patientsthereplacementwasfixedandwitha sat-isfactoryfunction.Thefourfailuresoccurredinpatientswith

thediffuseandactivetypeofthedisease,aspresentedinour case.Three failureswere duetoasepticloosening andone wasduetorelapseofthedisease.Theseauthorsrecommend abroadsynovectomy,associatedwithposteriorcruciate lig-amentreplacementforbettersynovialmembraneexposure, whichwasdoneinthiscase.

Total knee arthroplasty (TKA) in patients with VNS is not a complication-free procedure. In cases of recurrence ofthe disease afterTKA and dependingon functional sta-tus, a newsynovectomymay beattempted. Other options wouldberadiotherapy,arthrodesisorevenamputation.Many patientsalsohavedifficultyingainingrangeofmotion(ROM) intheinitialpostoperativeperiodandtheauthorsrecommend attentiontoavoidstiffness.10

TKA is a viable option for patients with DPVNS when thereisassociatedadvancedsecondaryosteoarthrosis.Itcan providepainrelieveandbetterlimbfunction.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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