Braz. j. . vol.81 número5

BrazJOtorhinolaryngol.2015;81(5):568---570

www.bjorl.org

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

CASE

REPORT

Lemierre

syndrome:

a

rare

complication

of

pharyngotonsillitis

夽

,

夽夽

Síndrome

de

Lemierre:

complicac

¸ão

rara

de

faringotonsilites

Hyun

Jin

Noh

_,

_Claudia

_Antunha

_de

_Freitas

_,

_Rafael

_de

_Paula

_e

_Silva

_Felici

_de

Souza

,

Juliana

Caminha

Simões

,

Eduardo

Macoto

Kosugi

a_{EscolaPaulistadeMedicina,UniversidadeFederaldeSãoPaulo(EPM-UNIFESP),SãoPaulo,SP,Brazil} b_{FaculdadedeMedicinadeSãoJosédoRioPreto(FAMERP),SãoJosédoRioPreto,SP,Brazil}

c_{Rhinology,EscolaPaulistadeMedicina,UniversidadeFederaldeSãoPaulo(EPM-UNIFESP),SãoPaulo,SP,Brazil}

Received3February2015;accepted16March2015 Availableonline21July2015

Introduction

Lemierre’ssyndromeconsistsofapharyngealinfection asso-ciatedwithsepticemiaandinternaljugularthrombosiswith septic emboli. This condition was described in 1936 by Andre Lemierre.1,2 _{It was initially named as post-anginal} septicemia, and then, ‘‘forgottendisease’’, by becoming arareconditionaftertheadventofantibiotics,withfewer than100casesreportedsince1974.2 _{Lamierre’ssyndrome} hasalsobeen namednecrobacillosis,due tothepresence

ofFusobacteriumnecrophorum,acommensalbacteriafrom

oralcavitywhichisconsideredthemostcommoncausative agentof the disease. Bacteroides,Streptococcus group B

and C, Streptococcus oralis, Staphylococcus epidermitis,

EnterococcusandProteusmirabilismayalsobeinvolved.3

夽 _{Pleasecitethisarticleas:NohHJ,deFreitasCA,deSouzaRPSF,}

SimõesJC,KosugiEM.Lemierresyndrome:ararecomplicationof pharyngotonsillitis.2015.BrazJOtorhinolaryngol.2015;81:568---70. 夽夽_{Institution:SectorofRhinology,Departmentof}

Otorhinolaryn-gologyand Head and Neck Surgery,Escola Paulistade Medicina, UniversidadeFederaldeSãoPaulo(EPM-UNIFESP),São Paulo,SP, Brazil.

∗_{Correspondingauthor.}

E-mail:edumacoto@uol.com.br(E.M.Kosugi).

Theobjectiveofthisreportis topresent a Lemierre’s-syndrome case treated at the Otorhinolaryngology Emer-gencyRoom.

Case

presentation

Malepatient,aged18years,presentedinitiallywiththroat painandleft-sideneck painevolvingwithfever(37.8◦_C).

This patient had been treated with benzathine penicillin andsymptomaticmedication.Onthe4thday,hebeganto experiencepaininhiskneesandankles.

Atadmission,thepatientwasinpoorgeneralcondition, with paleness, dehydration, tachycardia (116bpm), with 95% saturation in room air, hypotensive (108×57mmHg) and afebrile (36.6◦_{C).The patientpresented with}

hyper-emic oropharyngeal examination, a left cervical bulging, hepatomegaly and bilateral edema and hyperemia in his ankles.The initialinvestigation showedthrombocytopenia (28,000/mm3_{),leukocytosiswithaleftshift(16,100/mm}3_),

increasedcreatinine(3.5mg/dL)andurea(243mg/dL).The patient was treated according to the protocol for severe sepsis,includingbloodculture,vigoroushydration,IV cef-triaxone2g,andhospitalization.

Attheinvestigationforothersepticfoci,acervical ultra-soundscanshowedmultiplelymphnodeswithinflammatory reactionandthrombosisofupperandmiddlethirdsofleft

http://dx.doi.org/10.1016/j.bjorl.2015.03.009

Lemierresyndrome:ararecomplicationofpharyngotonsillitis 569

Figure1 (A)Rightcervicaldopplerultrasoundscanwithnormalvascularflow.(B)Flowabsentinleftinternaljugularvein.(C) Computedtomographyofchest;lungparenchymashowingmultiplenodes.(D)Ground-glassappearanceinlungbases.

internaljugularvein(Fig.1AandB).An ultrasoundof the lowerlimbsshowedasmallbilateralanklejointeffusion.

Duringthe1stdayofhospitalization,thepatient exhib-ited progressive dyspnea, with tachypnea, suprasternal depressionandstertorcrackesonpulmonary auscultation. We considered that the sepsis had its origin in a pul-monary focus, requiring tracheal intubation. Thoracic CT scan showed pleural effusion and multiple nodules dis-tributed through the lung parenchyma, with areas of a ground-glasspatterncorrespondingtosepticemboli(Fig.1C andD). Aftertheintroduction ofpiperacillin tazobactam, the patient’s evolution was favorable, with improvement ofthesepsis.The patientwasdischargedafter14daysof intravenous antibiotic therapy. Blood cultures showed no bacterialgrowth.

The initialfindingofpharyngealinfectionaccompanied bysepsis,incombinationwithconfirmationofjugularvein thrombosis and pulmonary septic emboli, validated the hypothesisofLemierre’ssyndrome.

Discussion

Lemierre’ssyndromeoccurspredominantlyinyoungadults1_; onestudyreportedhigherprevalenceinmen,4_asoccurred in this case. In most cases of this syndrome, a pharyngi-tisis notedbefore occurrence of systemiccomplications, similarly to what happened in our case; but the internal jugular thrombosis may result from other head and neck infections.5,6_{Theincidenceisestimatedtobe3.6casesper} millioninhabitants.2

The septic emboli originatingfrom the jugular throm-bosismainly affectlungsandjoints,3 _{exactlytheaffected}

fociin thecurrentreport.The lungconditionmayinvolve severe chest pain, dyspnea and hemoptysis due to pul-monaryabscesses. The joints aswell may develop septic arthritisandosteomyelitis.3

The treatmentof thissyndromeconsistsof supportfor the sepsis and antibiotic therapy, as was done in this case.Antibiotic therapy shouldbedirected toanaerobes, staphylococciandstreptococci. The main etiologicagent,

F.necrophorum,issensitive topenicillin,clindamycinand

metronidazole.6_{Thebloodcultureofourpatientshowedno} bacterialgrowth;butthereisaquestion:whetherthe previ-oususeofantibioticsorafailureinthecollectionprocedures foranaerobicorganismsmayhavehadsomeinfluenceonthe negativebacterialgrowthoutcome.

Final

considerations

Lemierre’ssyndromeisararediseasesecondarytocommon otorhinolaryngologicalinfections.Theotorhinolaryngologist shouldbealertforsignsofsepsisincommoninfectious con-ditions.Acorrectapproachtosepsisiscriticalforobtaining goodresultsinthissyndrome.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

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3.VargiamiEG,ZafeiriouDI.Eponym:theLemierresyndrome.Eur JPediatr.2010;169:411---4.

4.EykynSJ.Necrobacillosis.ScandJInfectDisSuppl.1989;62:41---6.

5.Goldenberg NA, Knapp-Clevenger R, Hays T, Manco-Johnson MJ. Lemierre’s and Lemierre’s-like syndromes in children: survival and thromboembolic outcomes. Pediatrics. 2005;116: e543---8.