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Neurological disease may precede lymphadenopathies in Actinomyces europaeus infection

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Pleasecitethisarticleinpressas:CorcioneS,etal.NeurologicaldiseasemayprecedelymphadenopathiesinActinomyceseuropaeus infection.JInfectPublicHealth(2017),https://doi.org/10.1016/j.jiph.2017.12.010

ARTICLE IN PRESS

G Model

JIPH-855; No.ofPages2

JournalofInfectionandPublicHealthxxx(2017)xxx–xxx

ContentslistsavailableatScienceDirect

Journal

of

Infection

and

Public

Health

j o u r n al ho me p ag e :h t t p : / / w w w . e l s e v i e r . c o m / l oc a t e / j i p h

Neurological

disease

may

precede

lymphadenopathies

in

Actinomyces

europaeus

infection

Silvia

Corcione

a,∗

,

Antonio

Curtoni

b

,

Ivana

A.

Paolucci

c

,

Giovanni

Di

Perri

a

,

Francesco

G.

De

Rosa

a,1

,

Rossana

Cavallo

b,1

aDepartmentofMedicalSciences,InfectiousDiseases,UniversityofTurin,Turin,Italy

bDepartmentofPublicHealthandPediatrics,MicrobiologyandVirologyUnit,UniversityofTurin,Italy

cDepartmentofClinicalandExperimentalMedicine,UnitofInfectiousDiseases,UniversityofMessina,Messina,Italy

a

r

t

i

c

l

e

i

n

f

o

Articlehistory: Received9October2017 Accepted7December2017 Keywords: Actinomyces Actinomycosis Vascularinfections Intensivecareunit Vasculitis

Neurologicalsymptoms

a

b

s

t

r

a

c

t

Actinomycesspeciesarepartofthecommensalfloraofthemucousmembranesoftheoropharynx, gastrointestinaltractandfemalegenitaltract.Actinomyceseuropaeusisashort,nonmotile,facultative anaerobicrodfirstdescribedin1997,susceptibleinvitrotoawiderangeofb-lactamantibioticswhich areregardedasfirstchoice.InthisreportwedescribedthecaseofA.europaeusinfectioninayoung femalepatientadmittedtoIntensiveCareUnitandthepossibledamageofvascularendotheliumdueto achronicprogressiveactinomycosisthatatfirstinvolvednecksofttissue,thencervicallymphnodes,and finallyextendedtothevascularstructure.

©2017PublishedbyElsevierLimitedonbehalfofKingSaudBinAbdulazizUniversityforHealth Sciences.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/

licenses/by-nc-nd/4.0/).

DearEditor,

Actinomycesspeciesarepartoforalmicrobiome,commensal floraofgastrointestinaltractandgenitaltractinwomen. Actino-mycesarehumanopportunisticpathogensandetiologicagentsof actinomycosisindifferentdistrictssuchasthecervicofacial, tho-racic,abdominalandalsothepelvicareasinwomen[1].

First described in 1997, Actinomyces europaeus is a short, nonmotile,Grampositiverod,facultativeanaerobicand suscep-tiblein vitrotoa widerange of b-lactamantibioticswhich are regardedasfirstchoice.SomeA.europaeushavereduced suscep-tibility totetracycline, ciprofloxacin, clindamycin, linezolid and piperacillin/tazobactam[2].

Wereportthecaseofa21-year-oldfemaleadmittedtoour hos-pitalon8thFebruary 2017afterthesuddenonsetof feverand headachefollowedbyhemiparesiswithseizures,requiring intu-bationandIntensiveCareUnitsupport.Herrecentmedicalhistory wasonlyconsistentwithsurgeryforaneartympanicorcutaneous abscessonJanuary2017,performedinanothercountrywithout anydischargenoteavailable.

∗ Correspondingauthor.

E-mailaddress:silvia.corcione@unito.it(S.Corcione).

1 Theseauthorsequallycontributedtothiswork.

Attime ofadmission, shewasempiricallytreated with van-comycinandmeropenemforasuspectedpost-surgicalmeningitis. Atphysicalexamination,thepatienthadaleftsidehemiplegia andatender,fluctuatingcollectionwasobservedontheright auri-cle,withasuspicionoffistulaauris:aswabwasperformedona densegranulomatoussecretionaftercompressionandsubmitted toMicrobiologylaboratoryforculture.

Acerebralcomputedtomography(CT)performedonFebruary 9threvealedarightsagittalandtransversussinusthrombosiswith subarachnoidhemorrhageintherightfrontallobeandastenosis oftheinternalcarotidartery.Becauseofanewepisodeofseizures, meropenemwasdiscontinuedafter3daysandceftazidimewas addedtovancomycin.

On February 11th sedation was suspended and the patient showeddysarthria,leftmotorhemisyndromeandanantalgic pos-ture, with left head deviation. Blood culture were persistently negative,rectalswabwaspositiveforcarbapenemaseproducingK. pneumoniaeandtherapywasempiricallyde-escalatedto trimetho-prim/sulfamethoxazole(TMP/SMX).

Forthepersistenceoftheleftheaddeviationandarightcervical lymphnodeswithtendernessandstiffnessoftherightsterno-cleido mastioidien observedat the physicalexamination, onFebruary 16thaCTscanwasperformed,showingrightretroauricularand jugularlymphnodes,multiplebilateralcervicallymphadenopathy andarightsideabscessinparotidregionwithsomehypodensitive colliquativeareas.

https://doi.org/10.1016/j.jiph.2017.12.010

1876-0341/©2017PublishedbyElsevierLimitedonbehalfofKingSaudBinAbdulazizUniversityforHealthSciences.ThisisanopenaccessarticleundertheCCBY-NC-ND license(http://creativecommons.org/licenses/by-nc-nd/4.0/).

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Pleasecitethisarticleinpressas:CorcioneS,etal.NeurologicaldiseasemayprecedelymphadenopathiesinActinomyceseuropaeus infection.JInfectPublicHealth(2017),https://doi.org/10.1016/j.jiph.2017.12.010

ARTICLE IN PRESS

G Model

JIPH-855; No.ofPages2

2 S.Corcioneetal./JournalofInfectionandPublicHealthxxx(2017)xxx–xxx

Fig.1. Actinomycesspp.isolatedfromtherightauricularfluctuatinglesion.

Fewdays later, a Gramstaining fromsmall circular smooth grayishcoloniesgrownonanaerobicculturemediaoffistulaauris swab,revealedGrampositiverods(Fig.1).Amatrix-assistedlaser desorptionionizationtime-of-flightmassspectrometryassay per-formedfromisolatedcolonyidentifiedA.europaeus withahigh degreeofconfidence.Theisolatewastestedforsusceptibilitywith thefollowingresults:penicillinMIC0.032␮g/mL;vancomycinMIC 0.25␮g/mL;clindamycinMIC>256␮g/mLandmetronidazoleMIC >256␮g/mL. Basedonmicrobiologicaland radiologicalfindings, TMP/SMXwasstoppedandaregimenwithvancomycinplus ampi-cillin/sulbactamwasstarted.Thepatientslightlyimprovedwitha reductionoftheparotidabscess,lymphadenopathyandastability ofherneurologicalfindings.Attimeoftransfertoalongtermcare facilities,theinfectiousdiseaseteamrecommendeda4weeks ther-apywithceftriaxoneintravenouslyfollowedbyoralamoxicillinfor atleast5months.

Actinomycesmaybepresentasnormalfloraintheoralcavity andpharynx,andtheirmostfrequentpresentationisanindolent, suppurativeprocess,mostcommonlyinthecervicofacialregion. Ourpatientunderwentdrainageoftympanicabscessonemonth beforetheclinicalonset,andthedisruptionofthemucosamight bethefrontdoorforthedevelopmentofActinomycesinfection.In ourcase,vascularinvolvementbyActinomyceswashypothesized atthelevelofarterialandvenoussystem.Sofar,onlythreecasesin literaturehavebeendescribedasanadjacentvasculatureextension fromcontiguousfoci[1,3].

Inthiscase,wemightspeculatethatthedamageofvascular endotheliummightbeduetoachronicprogressiveactinomycosis thatatfirstinvolvednecksofttissue,thencervicallymphnodes, and finally extended to the vascular structure. In fact, in an autopticreportofacerebralactinomycosis,theauthorsreported

an histologicalfinding of vasculitiswhich wasquite similarto thatobserved inpolyarteritis nodosa.Thecauseof polyarteritis nodosa-likevascular changes is unknown,but immune mecha-nismsappeartobeinvolvedandActinomycesmightbeoneofthe pathogenicagentsof polyarteritisnodosa-likevascular changes, aspreviouslyreportedfordifferentbacterialagents[4,5].

Inourcase, A.europaeus wasisolatedfromitsnormal niche asupperbodysiteanditwasfullysusceptibletopenicillin.Even thoughthe patientwasinitially treated withanactive therapy againstActinomyces,thenshewasstableandtherapywas empir-ically switched to TMX/SMT. Our patient did not receive any empiricaltherapycoveringactinomycosisforatleastoneweekand thismighthavepromotedtheprogressionofinfection.

Inconclusion,actinomycosisisarare,indolentandsometime aggressiveinfectionandcliniciansshouldbekeepanhighlevelof suspicionduetotheverydifferentrangeofclinicalmanifestations. Consent,forthepublicationforthiscasereportandany addi-tionalrelatedinformationwastakenfromthepatientinvolvedin thestudy. Funding Nofundingsources. Competinginterests Nonedeclared. Ethicalapproval Notrequired. Authorscontribution

SC,AC,FDRdraftedthepaper,SC,AC,IPcollectedclinicaland microbiologicaldata,SC,RC,FDR,GDPrevisedthepaper.

References

[1]Lezcano C, Simons JP, Colman KL, Cohen MS, Lin PL, Reyes-Mugica M. Actinomicotyc mastoiditis complicated by sigmoid sinus throm-bosis and labyrinthine fistula. Pediatr Dev Pathol 2014;17:478–81,

http://dx.doi.org/10.2350/14-05-1492-CR.1.

[2]SilvaWA,PinheiroAM,JahnsB,Bogli-StuberK,DrozS,ZimmerliS.Breastabscess duetoActinomyceseuropaeus.Infection2011;39:255–8.

[3]Koda Y, Seto Y, Takeichi S, Kimura H. Fatal subarachnoid hemorrhage complicatingactinomycotic meningitis. Forensic Sci Int 2003;134:169–71,

http://dx.doi.org/10.1016/j.nmni.2015.05.001.

[4]David J, Anseli BM, Woo P. Polyarteritis nodosa associ-ated with streptococcus. Arch Dis Child 1993;69:685–8,

http://dx.doi.org/10.1016/S0379-0738(03)00133-6.

[5]O’SullivanJ,MaurerB,TowersRP.Staphylococcusaureussepticaemia:theinitial presentationofpolyarterititsnodosa.BrJRheumatol1989;28:347–9.

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