Periprosthetic joint infection of a total hip arthroplasty with Candida parapsilosis

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CASE REPORT – OPEN ACCESS

InternationalJournalofSurgeryCaseReports69(2020)72–75

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

Periprosthetic joint infection of a total hip arthroplasty with Candida parapsilosis

Laurence Vergison

^a,b,∗

, Alexander Schepens

^a

, Koen Liekens

^a

, Renata De Kesel

^a

, Hans Van der Bracht

^a

, Jan Victor

^b

aDepartmentofOrthopaedicSurgeryandTraumatology,GeneralHospitalSt-Lucas,Ghent,Belgium

bDepartmentofOrthopaedicSurgeryandTraumatology,GhentUniversityHospital,Ghent,Belgium

a r t i c l e i n f o

Articlehistory:

Received9January2020

Receivedinrevisedform15March2020 Accepted18March2020

Availableonline31March2020

Keywords:

Fungalperiprostheticinfection Candidaparapsilosis

Surgicaltreatment Fluconazole Casereport

a b s t r a c t

INTRODUCTION:Fungalperiprostheticjointinfection(PJI)isadisruptiveandcomplexcomplicationof jointarthroplasty.WepresentacaseofafungalPJIwithCandidaparapsilosisafteratotalhiparthroplasty (THA).

PRESENTATIONOFCASE:A73-year-oldwomanwithahistoryofovariancancerwithperitonealmetas- tases,wastreatedwithaTHA,duetosymptomaticarthritisoftherighthip.Onemonthaftersurgery,she haddifﬁcultieswalking.Inﬂammatoryparametersweremildlyincreased.Aspirationofasubcutaneous abscessdiagnosedCandidaparapsilosis.Atwo-stagerevisionarthroplastywithoutspacerwasperformed.

Duringasix-weekprosthesis-freeinterval,intravenousﬂuconazole400mgwasgiven.Afterreimplan- tation,ﬂuconazolewascontinuedfortwoweeksintravenouslyandlife-longperorally.Follow-upofthe patientaftersixmonthsshowednorecurrenceofinfection.

DISCUSSION:ThiscaserevealedthatwhenPJIissuspected,alowtresholdforjointaspirationisimpor- tant.Two-stagerevisionwithsystematicantifungaltherapyisthepreferredtreatmentoffungalPJI.Our casedemonstratedagoodresultwithaprosthesis-freeinterval.Fluconazoleisthepreferredantifungal treatmentanditshouldbeappliedforatleastsixmonthsorlonger.

CONCLUSION:Toourknowledge,thisistheﬁrstcaseofafungalPJIwithCandidaparapsilosisafteraTHA treatedwithatwo-stagerevisionarthroplastywithoutspacerandalife-longﬂuconazoletreatment.

1. Introduction

Periprostheticjointinfection(PJI)is oneofthemostdisrup- tiveandcomplexcomplicationsofjointarthroplasty[1].Themost commonpathogensassociatedwithPJIareStaphylococcusspecies, whichareseenin50–60%ofallcases[1].

Fungalinfectionatthesiteofthejointreplacementisrareand isestimatedtoappearinapproximately1%ofallPJIs[2].Asthey canleadtodestructiveconsequencesifnottreatedtimely,fungal PJIisadiagnosticandtherapeuticchallenge[1,2].

WepresentacaseofCandidaparapsilosisearlyPJIinatotalhip arthroplasty(THA)ofa73-year-oldwoman.Atwo-stagerevision arthroplastywithoutspacerwasperformed.

Abbreviations: PJI,periprostheticjointinfection;CRP,c-reactiveprotein;ESR, erythrocytesedimentationrate;DTT,difﬁcult-to-treat;TKA,totalkneearthroplasty;

THA,totalhiparthroplasty;HPF,highpowerﬁeld;MSIS,MuskuloskeletalInfection Society;EBJIS,EuropeanBoneandJointInfectionSociety;IDSA,InfectiousDiseases SocietyofAmerica;DAIR,Debridement,antibiotics,irrigationandretention.

∗ Correspondingauthorat:DepartmentofOrthopaedicSurgeryandTraumatol- ogy,GeneralHospitalSt-Lucas,Groenebriel1,Ghent,Belgium.

E-mailaddress:laurence.vergison@skynet.be(L.Vergison).

Furthermore,acleardescriptionofthecurrentliteratureonthe riskfactors,clinicalfeaturesandtherapeuticstrategiesoffungalPJI ispresented.TheworkhasbeenreportedinlinewiththeSCARE criteria[3].

2. Presentationofcase

A 73-year-oldwoman, treatedfor ovarian cancerwithperi- tonealmetastases,wasknownwithsymptomaticarthritisofthe righthip.Shewastreatedconservativelywithintra-articularinfil- trations, because of active treatment of the peritoneal disease withBevacizumab (Avastin^®), a monoclonal antibodythat pre- ventswoundhealing.Duetotheheavyimpactofthehiparthritis onherqualityoflife,thepatientaskedforanoperativesolution, morespecificallyaTHA.Inconsultationwiththeoncologist,Beva- cizumabwastemporarilyinterrupted.Fourmonthsafterthethird intra-articularinfiltration,shewastreatedwithaTHA.TheTHAwas performedthroughadirectanteriorapproach.Acementlesstita- niumporouscoatedcupandacementlesstitaniumstraightstem withpolyethylenelinerandaceramicfemoralheadwereused.

Exactlyonemonthaftersurgery,shewasnotabletowalkwith- outpaininherrighthip,aftershewaspainfreetheﬁrsttwoweeks.

ApositiveTrendelenburgwaspresent.Hiprangeofmotion(ROM)

https://doi.org/10.1016/j.ijscr.2020.03.037

org/licenses/by/4.0/).

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L.Vergisonetal./InternationalJournalofSurgeryCaseReports69(2020)72–75 73

Fig.1.PlainX-rayshowsnoargumentsofprostheticloosening.

wasslightly reducedin internrotation. Therewasnoskinery- themaorsignsofsinustract.Shewasafebrile(T:36,3C)andhad nomalaise.Theradiographsshowednoargumentsofprosthetic loosening (Fig.1).TheC reactiveprotein(CRP) anderythrocyte sedimentationrate(ESR)weremildlyincreased,upto67mg/L(referencevalue:<5mg/L)and36mm/h(referencevalue:2–15mm/h) respectively.

Apercutaneousaspirationoftherighthipwasperformed,two daysafterthebloodresults.Duetoaninsufﬁcientamountofﬂuid, asecondaspirationbyultrasoundwasdone.Theultrasoundscan showedan extended subcutaneous abscess of 80mm long and 10mmdeep.BothculturesrevealedCandidaparapsilosis.

Atday40,theprosthesiswasremovedandthesurroundingtis- suesweredebrided(Fig.2).Atwo-stageexchangewithoutspacer andwithalongintervalwaschosen.Asix-weekcourseofintra- venousﬂuconazoleat400mgdailywasstarted.Sixweeksafter theremovalofherinitialprosthesis,arevisionTHAwasperformed throughapostero-lateralapproach.Acementlessdualmobilitycup and acementedtitaniumstraight stemwithpolyethylene liner anda ceramicfemoralheadwereused(Fig.3).Afterreimplan- tionofthehipprosthesis,theantifungaltherapywascontinued fortwoweeksintravenouslyandlife-longperorally.Follow-upof thepatientaftersixmonthsshowednorecurrenceofinfectionand apainfreemobilisationoftherighthip.

3. Discussion 3.1. Pathogenesis

Fungalinfectionaftertotaljointreplacementisrareandisseen inapproximately1%ofallPJIs[2].Themajorityoftheseinfections arecausedbyCandida(albicans,parapsilosis,glabrata,tropicalis), Aspergillus,CoccidioidesandBlastomycetes[2].

Overthepasttwodecades,theCandidaparapsilosispathogen is onthe riseworldwide. C. parapsilosis is knownto bea nor- malhumancommensal,consideringthefactthatitisoneofthe mostcommonlyisolatedfungifromthesubungalspaceofhuman hands.IncontrasttoC.albicansandC.tropicalis,priorcolonization isnotobligatepresentininfectionscausedbyC.parapsilosis,asitis usuallytransmittedhorizontallythroughhumanhands,prosthetic devices,medicalﬂuidsandcatheters[4].

Fig.2. PlainX-rayafterremovalofthetotalhiparthroplasty.Aprosthesis-free intervalof6weekswasperformed.

Fig.3. PlainX-rayafterrevisionofthetotalhiparthroplastywithdualmobilitycup andcementedstem.

The increaseof C. parapsilosis andother fungalPJI hasbeen attributedtoahigherriskinimmunosuppressedpatients,induced byunderlyingcausesincludingmalignantdiseases,drugtherapies, antibiotic overuse, indwelling catheters, diabetes, tuberculosis, multiplerevisionsurgeryandintravenousdruguse[4,5].

Someoftheseriskfactorswereseeninourcase,suchasmalig- nant diseaseand thepreviousdrugtherapy withBevacizumab, whichpreventswoundhealing.

As other microorganismssuch asrifampin-resistant staphy- lococci, enterococci and ciprofloxacin-resistant gram-negative bacteria,fungiare definedas difficult-to-treat(DTT)organisms.

Thesepathogensproducecomplexbioﬁlmswhichprovideresis-

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74 L.Vergisonetal./InternationalJournalofSurgeryCaseReports69(2020)72–75 Table1

DiagnosticcriteriaofPJIaccordingtotheEBJIS(2018).Atleastonethefollowing4criteriamustbefulﬁlled[9].

Diagnostictest Criteria Sensitivity Speciﬁcity

Clinicalfeatures Sinustractorvisiblepurulencearoundtheprosthesis 20–30% 100%

Leukocytesinsynovialﬂuid >2000/␮lleukocytesor≥70%granulocytes 93–96% 93–96%

Histology Inﬂammationinperiprosthetictissue(>2granulocytes/HPF) 95–98% 95–98%

Microbiology(culture)

Synovialﬂuidor 60–80% 97%

≥2periprosthetictissuesamples^aor 70–85% 92%

Sonicationﬂuid(≥50CFU/mL) 85–95% 95%

aForhighlyvirulentorganisms(e.g.Staphylococcusaureus,Escherichiacoli)onepositivetissuesampleissufﬁcienttoconﬁrminfection.

tancetobioﬁlm-activeantimicrobialsbylimitingthepenetration ofsubstancesthroughthematrix[6].

3.2. Clinicalfeatures

Patientsinfectedbyfungalorganismsdonotmandatorilyshow thesamesymptomsasthoseinfectedbybacterialorganisms.The symptomsareoftenindolentandcanvariatebetweenpain,ery- thema,swelling,anddecreasedmobility.Systemicsigns,suchas fever,chillsormalaisearerelativelyuncommon[7].Athorough evaluationofthepreviousincisionsisnecessary.Themeaninterval betweeninitialsurgeryandclinicalsignsis21months[6].

3.3. Diagnosis

AnearlydiagnosisofPJIisofmajorimportanceforpreserving theprosthesisandthejointfunctionality.CriteriafordiagnosisofPJI weredescribedbytheMuskuloskeletalInfectionSociety(MSIS),the EuropeanBoneandJointInfectionSociety(EBJIS)andtheInfectious DiseasesSocietyofAmerica(IDSA).Trampuzetal.describedthe EBJIScriteriaasthemostsensitivecriteriaforPJI(Table1)[8,9].

ThediagnosisofafungalPJIcanberatherchallenging.Incontrast tobacterialPJI,routineinflammatoryparametersarenotnecessar- ilyelevatedinfungalPJI[5].Anotherdifficultyinthediagnosisis themisinterpretationofpositivefungalcultureascontaminant.A fungalorganismintissueorfluidsampleobtainedbyaspiration ofaprostheticjointcanbeseenasagenuineinfectionorfungal colonization.Severalauthorsemphasizetheimportanceofmulti- plepositivetissueculturesbeforethediagnosisoffungalPJIcanbe made[5].However,accordingtootherauthors,onepositivesample mayconfirmthediagnosisofPJIincaseofhighlyvirulentorganisms [8,9].

3.4. Treatment

SeveraltreatmentoptionsforPJIhavebeendescribed,includ- ingdebridementwithpolyexchange,one- ortwo-stagerevision arthroplastyandchronicsuppressivetherapy[2].

Debridement,antibiotics,irrigationandretention(DAIR)isnot recommended astreatment for fungal PJI.Due tothe complex bioﬁlmformation,debridementalonewillleavefungalremnants atthesiteoftheinfection[5].

Atwo-stagerevisionisthepreferredtreatmentforfungalPJI.

Antifungaltreatmentshouldbeappliedbetweenexplantationand reimplantation for at least 6 weeks, as described by the IDSA [11].Fluconazoleisthepreferredantifungaltreatmentforfungal PJI.AmphotericinBlipidformulationsorechinocandinsareother optionsbutmaybelesstoleratedduetothesideeffects.Revision shouldbeperformedwhentherearenoclinicalsignsofinfection andbloodinfectionmarkershavenormalized.Followingreimpla- nation,dailytreatmentwithﬂuconazole400mg(6mg/kg)should bemaintainedfor6monthsorlonger,iftolerated[10].Somereports suggestatwo-weekantimicrobialholidaypriortoreimplantation, inordertocollecttruetissuespecimens forcultureatthetime

ofreimplantation[6].However,thereisnoconclusiveevidenceto supporttheantimicrobialholidayperiod[10].

Kuiperet al.analyzed 164patientswhohad fungalPJIafter TKAandTHAanddescribedasuccessrateof67/79(85%)inthose patientswhounderwentatwo-stagerevisioninafollow-upperiod ofmorethantwoyears[11].Otherauthorsreportederadication ratesof41%and50%aftertwo-stagerevision[1,12].

Zimmerlietal.proposedatwo-stageexchangewithoutaspacer asasuitabletreatmentfordifﬁcult-to-treatmicroorganisms[6].A temporaryantifungal-impregnatedspacerhasmultipleadvantages suchaspreservationofthejointspace,ahighlocalconcentrationof antifungaldrugsandaneasierreimplantationduetotheabsence ofscartissueinthemedullarycanalandintheacetabulum.How- ever,asspacersmaybeseenasaforeignbody,microorganismsmay adhereandcontinuetheinfection.Furthermore,variousmechani- calcomplicationswhenusingcementspacershavebeenreported suchasspacerfractures,dislocationsandfemoralfractures[13].

Antifungal treatment without surgery is not suggested and shouldonlybesupportedifthepatientrefusessurgeryorthesur- gicalprocedureisassociatedwithhighriskofthepatient’slife[8].

Inthiscase,chronicsuppressivetherapyafterreimplantationhas beenchosentopreventrelapse.

4. Conclusion

FungalPJIisadiagnosticandtherapeuticchallengeasitcanlead todestructiveconsequences[7].Weemphasizetheimportanceof alowthresholdforjointaspirationwhenPJIissuspected.

Two-stagerevisionwithsystematicantifungal therapyisthe preferredtreatmentoffungalPJIwitheradicationratesbetween 50%and93%[12].DAIRisnotrecommendedasatreatmentoption for fungal PJI, due tothe complex bioﬁlm formation of fungal pathogens[5].

Ourcasedemonstratedanearlyrecognitionandtreatmentof afungalPJIafterTHA.Atwo-stagerevisionwithoutspacerwas usedincombinationwith6weeksofﬂuconazoleintravenousdur- ingtheprosthesis-freeinterval.Afterreimplantation,ﬂuconazole wascontinuedfortwoweeksintravenouslyandlife-longperorally.

Aconsensusforthischallengingproblemislackingduetoinho- mogeneousstudycohorts,alimitednumberofcasereportsand caseseriesandthesmallpatientnumbers.Boththedurationofthe antifungaltherapyafterreimplantationastwo-stagerevisionwith orwithoutspacerremainquestionable.Futurehigh-qualitystudies arenecessarytoaccuratelymanagefungalPJI.

DeclarationofCompetingInterest

Theauthorsdeclarenoconﬂictofinterest.

Sourcesoffunding

Therewerenosourcesoffunding.

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CASE REPORT – OPEN ACCESS

L.Vergisonetal./InternationalJournalofSurgeryCaseReports69(2020)72–75 75 Ethicalapproval

Ethicalapprovalwasnotrequiredbyourinstitution.

Consent

Thepatientdescribedinthiscasereportgaveherinformedcon- sentfortheinclusioninthispublication.

Authorcontribution

LaurenceVergison:writing-originaldraft,reviewandediting, visualisation.

AlexanderSchepens:writing-revisingandediting,supervision, responsiblefortreatmentprotocol.

Koen Liekens: resources, investigation, surgeon of the case, revisingthemanuscript.

RenataDeKesel:revisingthemanuscript.

HansVanderBracht:revisingthemanuscript,generalscientiﬁc coordinator.

JanVictor:revisingthemanuscript,supervision.

Registrationofresearchstudies Notapplicable.

Guarantor

LaurenceVergison.

Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed.

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