Rev. bras. ortop. vol.51 número2

r e v b r a s o r t o p . 2016;51(2):231–234

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

Case

Report

Phaeohyphomycosis

infection

in

the

knee

夽

David

Sadigursky

_,

_Luisa

_Nogueira

_e

_Ferreira,

_Liz

_Moreno

_de

_Oliveira

_Corrêa

HospitalCOT,Salvador,BA,Brazil

a

r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received22March2015 Accepted28April2015

Availableonline23February2016

Keywords:

Infection Fungi Knee

Dermatomycoses

a

b

s

t

r

a

c

t

Phaeohyphomycosisiscausedbycutaneousfungiandrarelyaffectslargejoints.Thisisa casereportonphaeohyphomycosisintheleftkneeofanelderlyindividualwithout immuno-suppression.Itwasaccompaniedbypainandswellingtheanteriorknee.Thecasewasfirst suspectedtobesuprapatellarbursitis,andwastreatedwithnonsteroidalanti-inflammatory drugs,withoutremissionofsymptoms.Surgicaltreatmentwasperformed,withresectionof thesuprapatellarbursaandanteriorregionofthequadricepstendon.Thematerialwassent foranatomopathologicalexaminationandculturing.Thepathologicalexaminationshowed phaeohyphomycosis. Thetreatmentinstitutedconsistedofitraconazole,200mg/dayfor sixweeks,andcompleteremissionofsymptomswasachieved.Thephysicalexamination remainednormalafteroneyearoffollow-up.Thisisthefirstpublishedcaseof phaeohy-phomycosisinfectioninthesuprapatellarregionoftheknee.Althoughalmostallthecases reportedhavebeenassociatedwithimmunosuppressedpatients,thiswasanexception.It isimportanttosuspectphaeohyphomycosisincasesofkneeinfection,intheareaofthe suprapatellarbursa,whenthesymptomsdonotresolveafterclinicaltreatment.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublishedbyElsevierEditora Ltda.Allrightsreserved.

Infecc¸ão

por

feohifomicose

em

joelho

Palavras-chave:

Infecc¸ão Fungos Joelho

Dermatomicoses

r

e

s

u

m

o

Afeohifomicose,causadaporfungosdemáceos,raramenteacometegrandesarticulac¸ões. Esteé um relatode casode feohifomicose,em joelhoesquerdode idoso não imunos-suprimido, acompanhado de dor e aumento de volume em regiãoanterior do joelho. Suspeitou-sedebursitesuprapatelar,sendomedicadocomanti-inflamatórionãoesteroidal, semapresentarremissãodossintomas.Fez-setratamentocirúrgico,foramressecadasa bursasuprapatelarearegiãoanteriordotendãodoquadrícepssendoapec¸aencaminhada

夽

WorkperformedintheCOTHospital,Salvador,BA,Brazil. ∗ _{Correspondingauthor.}

E-mail:davidsad@gmail.com(D.Sadigursky). http://dx.doi.org/10.1016/j.rboe.2016.02.004

232

paraexameanatomopatológicoecultura.Noexameanatomopatológicofoipossível eviden-ciarodiagnósticodefeohifomicose.Otratamentoinstituídofoiitraconazol,200mg/diapor seissemanas,apresentandoremissãocompletadoquadro.Oexamefísicosemanteve nor-malapósumanodeseguimento.Esteéoprimeirocasopublicadoarespeitodainfecc¸ãopor feohifomicoseemregiãosuprapatelar.Apesardequasetodososcasosregistradosestarem associadosapacientesimunossuprimidos,estefoiumaexcec¸ão.Éimportantequese sus-peitedefeohifomicosenasinfecc¸õesdejoelho,naáreadabursasuprapatelar,quandoos sintomasnãoresolveremapósotratamentoclínicomedicamentoso.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublicadoporElsevier EditoraLtda.Todososdireitosreservados.

Introduction

Phaeohyphomycosis isthe term used todescribe infection causedbyvariousspeciesofdematiaceousfungiwith black-ishpigment,especiallyofthegeneraWangiella,Alternariaand

Exophiala.1–3

Itisfoundinwidelydistributedinsoilaroundtheworld.It isanuncommoncauseofillnessamonghumans,butmay cause infections inboth immunosuppressedand immuno-competentindividuals.4 _{Almostallthe casesreportedhave}

beenassociatedwithimmunosuppressedpatientswhohave undergoneorgantransplantation orpatientstreateddueto malignantgrowths.5

Cutaneouslesionsandsubcutaneousnodulesarethe com-monestformsofpresentation.Mostinfectionsaresuperficial andareprecededbylocaltrauma.6_{Thisdiseaseisonlyvery}

rarelyassociatedwithinfection injoints:onlyasinglecase hasbeenreportedsofar,whichwasassociatedwithtearing ofaflexortendon.7

Here,wereportacaseofphaeohyphomycosisintheleft kneeofapreviouslyhealthypatientwhowasnot immuno-suppressed.

Report

of

clinical

case

Thepatientwasa76-year-oldmanwithahistoryofpainand edemainhisleftkneeforaroundtwomonths,withincreased volumeintheanteriorregionofthepatella.

Whenhewasfirstseen,hepresentedpainandincreased volumeintheanteriorregionofthekneeandsuprapatellar bursitiswassuspected.Hewasmedicatedwithanonsteroidal anti-inflammatorydrug.Hecamebacksevendayslater,with increasedvolumeand pain. Thesite waspunctured and a thicksecretionofpurulentcolorationwithyellowishlumps was observed. He was medicated with an oral antibiotic becauseofsuspectedsuperficialinfection.However,hecame back twodays later with recurrenceof the edemaand an increasedstateofpain.

Thepatient wasadmitted tohospitalforsurgical treat-mentconsistingofdrainage,resectionandcleaning.During the operation,thick materialofwhitish and yellowish col-orationwasobservedinalargequantityoffriablesolidlumps. Thesuprapatellarbursaandallthematerialintheanterior regionofthequadriceps tendonwasresected andsent for

Fig.1–Anatomopathologicalexamination.

Fig.2–Anatomopathologicalexamination.

anatomopathologicalexaminationandculturingonfour sam-ples.

Theresultfromthecultureswasnegative.However,the anatomopathological analysisshowed phaeohyphomycosis, asdemonstratedinFigs.1–4.

rev bras ortop.2 0 1 6;51(2):231–234

233

Fig.3–Anatomopathologicalexamination.

Fig.4–Anatomopathologicalexamination.

Thelaboratorytestsshowed:

• Anti-Histoplasmaantibodies:negative

• Hb:14.1

• Leukocytes:8800

• Platelets:182,000

• VHS:7

• Urea:26;creatinine0.9

• TGO:17.3;TGP:15.4;GGT:14.5;FA:139

• LDH:281.9

• PPD:unreactive

The screening tests for inflammatory arthritis and immunosuppressive diseases were confirmed as normal. Regardingthepatient’shabits,hewasnotasmoker,alcohol abuserordruguser.

Thephysicalexaminationremainednormalafteroneyear of follow-up, will full weight-bearing and complete range of motion. He was able to do routine activities without limitations.

Discussion

The clinical casepresentedhere is the first onepublished sofar oninfectionduetophaeohyphomycosisintheknee. The infection was located in the suprapatellar region and resembledbursitis.Inthiscase,thepatientdidnothaveany immunosuppressivediseases,unlikeinthecasepresentedby Chahaletal.,7_{inwhichtheinfectionledtotearingoftheflexor}

tendon.Moreover,thepatienthadHIV,wasanalcoholicand asmoker.

Data on the incidence of phaeohyphomycosis are still scarceintheliterature.InastudyinSanFrancisco(USA),the incidencewas1:1,000,000peryear.8

Theinfectionresultsfromdirectinoculationaftertrauma, cutsorwoundsthatarecontaminatedwithearth,vegetation ordecomposingwood.

Althoughalmostallthecasesrecordedhavebeen associ-atedwithimmunosuppressedpatientswhohad undergone organ transplantation or procedures to treat malignant growths, the presenceof immunosuppressionisnot oblig-atory for the disease to appear, as was seen in our case.2,3,5,9

Thelocationaffectedispreferentiallytheupperandlower limbs.Theneck,faceandbuttocksarerarelyaffected.10

In a study on 72 patients, 76% presented fever and 33% cutaneous manifestations, including skin eruptions and ulcers. Thecommonest siteof infection has been the lungs, which was seen in 33 (46%) of the 72 patients. This was followed by the heart, in 21 patients (29%); skin, in 19 (26%); brain, in 16 (22%); and kidneys, in 16 (22%).Theliver,spleen,lymphaticganglia,bones,jointsand muscles have been less commonly reported as infection sites.5

Thecaseofphaeohyphomycosisinthekneereportedhere isconcordantwiththeliterature,inthatcaseslocatedinjoints arerare.

Totreatphaeohyphomycosis,drugssuchasitraconazole, ketoconazole, fluconazole and flucytosine have frequently beenused.5_{Itraconazoleisthepreferreddrug.}10_Inonestudy,

thedrugmostusedwasamphotericinB,whichwas admin-isteredto62(97%)ofthe64patientswhoreceivedantifungal therapy.5

Rare fungiofsoilshavepathogenic potential inchronic skinlesionsandleadtoformationofcrustsandulceration.In patientswhohavereceivedtransplants,theyneedtobetaken into consideration inthe differential diagnosis forinvasive fungalinfections,inimmunocompromisedpatients.11

It is important to maintain a high degree of suspicion of phaeohyphomycosis in cases of infected tenosynovitis whenthesymptomsdonotbecomeresolvedafterirrigation, debridementandantibiotictherapy.7

Furtherstudiesonphaeohyphomycosisinjointsshouldbe conductedbecauseofthescarcityofsuchinformation.

Conflicts

of

interest

234

r

e

f

e

r

e

n

c

e

s

1. FerreiraLM,PereiraRN,DinizLM,SouzaFilhoJB.Casopara diagnóstico.AnBrasDermatol.2006;81:291–3.

2. CunhaFilhoRR,SchwartzJ,RehnM,VettotatoG,Resende MA.FeohifomicosecausadaporVeronaeabothryosa:relatode doiscasos.AnBrasDermatol.2005;80:53–6.

3. SilveiraF,NucciM.Emergenceofblackmouldsinfungal disease:epidemiologyandtherapy.CurrOpinInfectDis. 2001;14(6):679–84.

4. RevankarSG.Phaeohyphomycosis.InfectDisClinNorthAm. 2006;20(3):609–20.

5. RevankarSG,PattersonJE,SuttonDA,PullenR,RinaldiMG. Disseminatedphaeohyphomycosis:reviewofanemerging mycosis.ClinInfectDis.2002;34(4):467–76.

6. ChowdharyA,MeisJF,GuarroJ,deHoogGS,KathuriaS, ArendrupMC,etal.ESCMIDandECMMjointclinical guidelinesforthediagnosisandmanagementofsystemic phaeohyphomycosis:diseasescausedbyblackfungi.Clin MicrobiolInfect.2014;20Suppl3:47–75.

7.ChahalJ,DhotarHS,AnastakisDJ.Phaeohyphomycosis infectionleadingtoflexortendonrupture:acasereport. Hand(NY).2009;4(3):335–8.

8.ReesJR,PinnerRW,HajjehRA,BrandtME,ReingoldAL.The epidemiologicalfeaturesofinvasivemycoticinfectionsinthe SanFranciscoBayarea,1992–1993:resultsof

population-basedlaboratoryactivesurveillance.ClinInfect Dis.1998;27(5):1138–47.

9.DuvicM,LoweL,RiosA,MacDonaldE,VanceP.Superficial phaeohyphomycosisofthescrotuminapatientwiththe acquiredimmunodeficiencysyndrome.ArchDermatol. 1987;123(12):1597–9.

10.RossettoAL,DellatorreG,PérsioRA,RomeiroJCM,CruzRCB. FeohifomicosesubcutâneaporExophialajeanselmeilocalizada nabolsaescrotal–Relatodecaso.AnBrasDermatol. 2010;85(4):517–20.

11.GordonRA,SuttonDA,ThompsonEH,ShrikanthV,Verkley GJ,StielowJB,etal.Cutaneousphaeohyphomycosiscausedby Paraconiothyriumcyclothyrioides.JClinMicrobiol.