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BrazJOtorhinolaryngol.2015;81(1):115---116

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

www.bjorl.org

CASE

REPORT

Lemierre’s

syndrome:

a

pharyngotonsillitis

complication

,

夽夽

Síndrome

de

Lemierre:

uma

complicac

¸ão

de

faringotonsilite

Pedro

Ernesto

Barbosa

Pinheiro

,

Priscilla

Durante

Miotto,

Natalia

Quinhone

Shigematsu,

Edwin

Tamashiro,

Fabiana

Cardoso

Pereira

Valera,

Wilma

Teresinha

Anselmo-Lima

DepartmentofOphthalmology,OtorhinolaryngologyandHeadandNeckSurgery,SchoolofMedicineofRibeirãoPreto,University ofSãoPaulo,RibeirãoPreto,SP,Brazil

Received21September2014;accepted17October2014 Availableonline21November2014

Introduction

Lemierre’ssyndrome(LS)isarareandserious pharyngoton-sillitiscomplicationthatusuallyoccursinadolescentsand youngadultscausedbyanaerobicbacteria,morespecifically attributedtoFusobacteriumnecrophorum.Firstdescribed intheearlytwentiethcentury,itwasin1936thattheFrench microbiologist,Dr.AndréLemierre,outlinedthe character-isticsofthedisease.1

Case

report

Afemalepatient(K.C.O.),12yearsold,previouslyhealthy,

presented withsore throatand fever for seven days.She

wasdiagnosedinanothercenterwithpharyngotonsillitisand

receivedintramuscularBenzathinebenzylpenicillin,

evolv-ingwithpainimprovement,butwiththeonsetofpersistent

Pleasecitethisarticleas:PinheiroPE,MiottoPD,Shigematsu NQ, Tamashiro E, Valera FC, Anselmo-Lima WT. Lemierre’s syn-drome:apharyngotonsillitiscomplication.BrazJOtorhinolaryngol. 2015;81:115---6.

夽夽

Institution:FaculdadedeMedicinadeRibeirãoPretoda Uni-versidadedeSãoPaulo(USP),RibeirãoPreto,SP,Brazil.

Correspondingauthor.

E-mail:pedroernesto.fmrp@gmail.com(P.E.B.Pinheiro).

high temperature and bulging in the right anterior

cervi-calregionfor threedays.Hyperemiawasobserved inthe

oropharynxandcervicalbulgingontopographyoftheright

sternocleidomastoidmuscle,painfulonpalpation.Contrast

tomographyandangiographyshowrightperitonsillar

inflam-mation,thrombosisofrightinternaljugularvein(IJV),and

pulmonarynodulesconsistentwithmicroabscesses(Fig.1).

Initially,thediagnosisofLSwasnotconsidered,and

intra-venousantibiotictherapywasstartedwithamoxicillinand

clavulanate at a dose 90mg/kg/day, and anticoagulation

withenoxaparinandwarfarin,withgradualclinical

improve-ment.Afterthreedays,withthefinaldiagnosisestablished

andinconjunctionwiththeHospitalInfectionControl

Com-mittee,weoptedtokeeptheregimenduetoimprovement

inclinicalandlaboratoryparameters.Evenwiththepatient

being admitted on antibiotics, samples for general and

anaerobic bacteria cultures were harvested; however, no

bacterialgrowthwasobserved.Thepatientwasdischarged

after14 days of hospitalization, asymptomatic, with

pre-scribed oral antibiotics tocomplete 21 daysof treatment

andoralanticoagulationtherapy.Afterfourmonthsof

anti-coagulationtherapy,a controlresonanceangiography was

performedtomonitor thedisease, showingpersistenceof

IJV thrombosis. The patient remains on anticoagulation

medicationonregularclinicalvisitsbasis,withfollowupby

Otolaryngology,PediatricHematologyandVascularSurgery

teams.

http://dx.doi.org/10.1016/j.bjorl.2014.10.004

(2)

116 PinheiroPEetal.

Figure 1 Left, Cross-Sectional Computed Tomography showing pulmonary microabscesses. Right, Post-contrast Sagittal T1-weightedMagneticRessonanceAngiography,showingtheRightInternalJugularVeinfillingfailure.

Discussion

F. necrophorum is a Gram-negative anaerobic bacteria of

theoropharyngeal floracapable of causing primary

infec-tioninhealthyindividualswithintactanatomicalbarriers.

Thecondition begins witha sorethroatandprogresses to

impairment of peritonsillar tissue, parapharyngeal space,

IJVthrombophlebitisand,finally,septicemboliformation.2

The onset of sepsis occurs with marked fever (39---41◦C,

102---106◦F),typically4---5daysafterthestartofsorethroat

and sometimes after improvement of the symptom.2 The

appearanceofthejawanglebulging,orparalleltothe

stern-ocleidomastoidmuscle,clinicallyreflectsthrombophlebitis.

Lungsaretheprimarysiteof abscessformation secondary

to septic emboli; however, one can find multiple organ

involvement.2,3 LS diagnosis involves: (1) recent

orophar-ynx infection; (2) clinical or radiological evidence of IJV

thrombosis;(3)isolationofanaerobicpathogens;(4)atleast

one septic focus.4 There is controversy in the literature

regardingtheclassificationofcasesinwhichthepathogen

cannotbeisolated.2,3 However,considering thatthereare

negativeculturesin12%ofthecases,3facedwiththeclinical

conditionandthedifficultyofbacterialgrowthinpatients

duringantibiotictherapy,wemaintainedthediagnosisand

management in this case. Treatment includes antibiotics

for 3---6 weeks, with coverage for anaerobes.5

Anticoagu-lationtherapyiscontroversial,butitiswidelyused.Inthe

absenceof contraindications,thetherapymustbe

consid-ered, especiallyin patients withpoor clinical responsein

spiteofantibiotic.6

Final

comments

Aftertheadventofantibiotics,casesofLSpractically

dis-appeared,butthereis evidenceofitsincreasedincidence

in recent years, possibly associated with reduced use of

antibioticsforsorethroat.DescribedbyAndréLemierreas

a‘‘syndromethatissocharacteristicthatmistakeisalmost

impossible’’,1thisconditionispotentiallyasseriousasitis

potentiallytreatable.3Ahighdegreeofsuspicioniscrucial

toavoid a delayed diagnosis with potentially fatal

conse-quences.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

1.LemierreA.Oncertainsepticaemiasduetoanaerobicorganisms. Lancet.1936;1:701---3.

2.BaigM,RasheedJ,SubkowitzD,VieiraJ.Areviewoflemierre syndrome.InternetJInfectDis[serialonline].2005;5.Available from:http://ispub.com/IJID

3.RiordanT,WilsonM.Lemierre’ssyndrome:morethanahistorical curiosa.PostgradMedJ.2004;80:328---34.

4.AsnaniJ,JonesS.Casereview.JFamPract.2014;63:193---6. 5.KarkosPD,AsraniS,KarkosCD,LeongSC,TheochariEG,

Alex-opoulou TD, et al. Lemierre syndrome: a systematic review. Laryngoscope.2009;119:1552---9.

Imagem

Figure 1 Left, Cross-Sectional Computed Tomography showing pulmonary microabscesses. Right, Post-contrast Sagittal T1- T1-weighted Magnetic Ressonance Angiography, showing the Right Internal Jugular Vein filling failure.

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