Braz. j. . vol.83 número6

BrazJOtorhinolaryngol.2017;83(6):726---729

www.bjorl.org

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

CASE

REPORT

Bilateral

parotid

glands

infection

caused

by

Calmette-Guerin

Bacillus

after

intravesical

therapy

for

recurrent

bladder

cancer:

a

case

report

夽

Infecc

¸ão

bilateral

da

glândula

parótida

causada

pelo

bacilo

de

Calmette-Guérin

após

terapia

intravesical

para

câncer

recorrente

de

bexiga:

relato

de

caso

Eviatar

Friedlander

,

Paula

Martínez

Pascual

,

Pedro

Montilla

de

Mora

,

Bartolomé

Scola

Yurrita

a_{GregorioMara˜nónGeneralUniversityHospital,DepartmentofOtorhinolaryngology,Madrid,Spain}

b_{GregorioMara˜nónGeneralUniversityHospital,DepartmentofMicrobiologyandInfectiousDisease,Madrid,Spain}

Received30September2015;accepted11October2015 Availableonline6February2016

Introduction

The Calmette---Guerin Bacillus (BCG), an attenuated live strainofMycobacteriumboviswasoriginallydevelopedby AlbertLeonCharlesCalmetteandCamileGuerinin1925as a vaccine for tuberculosis.At the present, BCG is widely used as a complementary therapy for superficial bladder neoplasms. This treatment is considered to be safe and welltoleratedalthoughlocalanddistalcomplicationsmay appear.1 _{In 2003,Diaz etal. reportedin the Spanish}

lan-guage,for the firsttime,acase of unilateral infectionof theparotidglandcausedbyBCG.2_{WereviewedtheEnglish}

literaturefrom1975 to2015 andfound no other casesof such infection,as it is extremely rare. In this article we reportawell-documentedcaseofabilateralinfectionofthe

夽 _{Pleasecitethisarticleas:FriedlanderE,PascualPM,deMoraPM,}

YurritaBS.Bilateralparotidglandsinfectioncausedby

Calmette-Guerin Bacillus after intravesical therapy for recurrent bladder

cancer:acasereport.BrazJOtorhinolaryngol.2017;83:726---9.

∗_{Correspondingauthor.}

E-mail:[email protected](E.Friedlander).

PeerReviewundertheresponsibilityofAssociac¸ãoBrasileirade

OtorrinolaringologiaeCirurgiaCérvico-Facial.

parotidglandsproducedbyBCGanddiscussthetreatment andpatientfollow-up.

Case

report

Wepresent thecase ofa72-yearsoldmalewithahistory of recurrent bladder carcinoma in the last three years. Thepatientalsohadalonghistoryoftobaccoconsumption in the past 30years.He wasreferred toour ENT depart-ment due tobilateral parotid glandswellingwitha small ulcerativelesionontherightglandthathadbeen growing in the previous month. The patient underwent several treatments in the last few years for a recurrent bladder carcinoma.Thelasttreatmentconsistedofatransurethral resectionfollowedbyintravesicalinstillationtherapywith BCG. Treatment ended two month prior to the patient’s consultationwithourENTdepartment.The initialphysical examinationrevealed enlargementofbothparotidglands, with an elastic consistency. On the inferior pole of the rightglandtherewasasmallpainlessulcerwithcontinuous pus discharge (Fig. 1). Blood work showed mild anemia with no other findings. An echography-guided puncture of the right lesion was realized and samples were sent for microbiologic testing. An urgent CT scan was then

http://dx.doi.org/10.1016/j.bjorl.2015.10.017

1808-8694/©2016Associac¸˜aoBrasileiradeOtorrinolaringologiaeCirurgiaC´ervico-Facial.PublishedbyElsevierEditoraLtda.Thisisanopen

BilateralparotidglandsinfectioncausedbyCalmette-GuerinBacillusafterintravesicaltherapy 727

Figure1 Bilateralswellingofthe parotidglands. Theright parotid glandshows asmall,painless ulcerwith continuous pus discharge.

Figure 2 CT coronal section showing multiple trabecular zoneswithcysticandnecroticareas,seenbetterontheright sideinthisimage.

preformedshowingalargemassof6.4cm×3cm×3.6cm, on the right side. The lesion showed multiple trabecular zoneswithcysticandnecroticareas.The leftsideshowed a 3cm×2cm×2cm lesion with the same characteristics (Fig.2).Consideringage,sexandthe smokinghistory the preliminarysuspicionwasabilateralWarthin’stumor.Based

ontheanalysismadebythemicrobiologydepartmentusing GeneXpert®_{(Cepheid),theinitialreportcamebackpositive}

forMycobacterium tuberculosiscomplex.Giventhe result andthehistoryofoncologicbladdertreatmentswithBCG, molecular biology procedures (GenoType Mycobacterium CM/AS®_{,Hain)wereperformedconfirmingthepresenceof}

theattenuatedstrainofM.bovis,thusBCG.Ascintigraphy wasperformedtoruleoutotherlocationsofdissemination oftheBCG.Vertebral,renal andpulmonarydissemination areas were found. Patient underwent treatment with Isoniazid 300mg withvitamin B6, ethambutol800mgand levofloxacin 500mg once daily for 10 month presenting excellent response to the treatment. After 20 month of follow-uppatientstillhadnoevidenceofdisease,confirmed by CT scans and fiscal examination and blood tests. The swellingof the parotid glands completely disappeared as wellastheulcerativelesionontherightside(Fig.3).

Discussion

Tuberculosisaffectingsalivaryglandsisararetypeof extra-pulmonar tuberculosis, being the parotid gland the most frequent locationin thesecases. This entity maybe mis-diagnosedasa neoplasm, asthe clinical courseis usually thesame:alocalizedandprogressive, chronicswellingof thegland.Sometimesitcanevencausefacialpalsy, mim-ickingamalignanttumor.Inourcasethebilateralaffection inaneldersmoker manshouldincludeWarthin’stumor as afrequent possible diagnosis, asit appears asapainless, cystic,slow-growingmasswhichmainlyaffectstheparotid gland.In15%ofcasesitcaninvolvebothglands,anditdoes notusuallybecomemalignant.

728 FriedlanderEetal.

Figure3 Picturesobtainedaftertreatmentwascompleted(10month).Theparotidglandswellingreducedonbothsidesandthe ulcerativelesionontherightsidehealed.

techniqueto differentiate both types of pathologies. His-tological and microbiological tests must be run on the tissuesample.Histologytestingoftenshowsgranulomatous inflammation,whilethemicrobiologicaldiagnosiscouldbe achievedbyacid-fastbacillistaining,mycobacterialculture orPCR-basedassays.Unfortunately,culturesresultnegative in40.9%3_{of cases,duetothe controlofbacillary}

replica-tion by an immunocompetent host against an attenuated mycobacteria strain. A CT scanmay be a helpful tool in achievingthecompletediagnosisasithelpsdeterminethe extensionofthediseaseanditisusedinthefollow-up to evaluatetheresponsetothetreatment.2

Incasesinwhichtheecography-guidedpuncturecomes upwithnoclearresult,parotidectomywithintraoperative biopsycouldbedonetoruleoutmalignancy.2

The parotid tuberculosis is thought to be caused by hematogenousorlymphaticdisseminationofthebacillifrom the lungs or secondary to an oral cavity infection with involvementoftheStenon’sductandtheafferentlymphatic vessels.

In our case the patient didnot have any contact with

M.tuberculosis,butwithanattenuatedstrainofM.bovis, theBCG.Despiteitsrareincidence,thehematogenous dis-seminationof the bacilli is possible, and some conditions likeimmunosuppression,disruptionoftheurothelialbarrier duetotraumaticcatheterization,concurrenturinarytract infectionor earlyinstillationafter transurethral resection maypromoteit.3_{The disseminatedBCGinfectionisa}

dif-ferentconditionfromtheflu-likesymptomspost-instillation andthelocalcomplicationsinthegenitourinarytractthat canappearduringtreatment.Itisdefinedasapositiveblood orbonemarrowcultureorevidenceofinfectionattwoor more anatomicsites beyondthe urinary tract.3 _{The sites}

mostcommonly affectedare thelungs and the

osteoarti-cular system,3 _{beingthesalivaryglands anextremely odd}

locationfortheBCGtomigrate.

Froma therapeutic point of view, this disease can be treatedasanyothertuberculosisinfectionwith antitubercu-losisdrugs.TheBCGisintrinsicallyresistanttopyrazinamide andcycloserine2,3_{sothepreferredtreatmentisa}

combina-tionof isoniazid,rifampicin or ethambutol.Insome cases theBCGstrainissusceptibletoquinolonessoanagentfrom thisgroupmaybeaddedaswell.

Conclusions

The dissemination of BCG to the parotid gland is an extremely rarecondition. Incases of bilateralswelling of theparotidglandsinelderlypatientswithalonghistoryof smoking, bilateral Warthin’stumor maybe theinitial sus-picion. Adetailedhistoryof thepatientmustbeobtained toachieve differentialdiagnosisand ruleoutall possibili-ties.Theuseofechography-guidedpuncturesandmolecular biologyprocedureswasfundamentalinachievingthe diag-nosisinthiscase.Aninterdisciplinaryapproachandagood collaborationspecialistareessentialinthemanagementof complexescasessuchasthisone.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

BilateralparotidglandsinfectioncausedbyCalmette-GuerinBacillusafterintravesicaltherapy 729

2.DíazC,BaldóC,MartínA,FernándezMJ,Mu˜nozM,RodríguezL, etal.ParotidtuberculosisfollowingintravesicalBCGinstillation: acasereport.ActaOtorrinolaringolEsp.2003;54:129---33.

3.Pérez-Jacoiste Asín MA, Fernández-Ruiz M, López-Medrano F, Lumbreras C, Tejido A, San Juan R, et al. Bacillus