Isolation of Coxiella burnetii from an acromioclavicular infection with low serological titres

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Isolation of Coxiella burnetii from an acromioclavicular

infection with low serological titres

Clea Melenotte, Geraldine Bart, Francoise Kraeber-Bodere, Serge Cammilleri,

Benoit Le Goff, Didier Raoult

To cite this version:

Clea Melenotte, Geraldine Bart, Francoise Kraeber-Bodere, Serge Cammilleri, Benoit Le Goff, et

al.. Isolation of Coxiella burnetii from an acromioclavicular infection with low serological titres.

International Journal of Infectious Diseases, Elsevier, 2018, 73, pp.27-29. �10.1016/j.ijid.2018.05.018�.

�hal-01858909�

Case

Report

Isolation

of

Coxiella

burnetii

from

an

acromioclavicular

infection

with

low

serological

titres

Cléa

Melenotte

,

Géraldine

Bart

,

Francoise

Kraeber-Bodere

,

Serge

Cammilleri

,

Benoit

Le

Goff

,

Didier

Raoult

*

a

Aix-MarseilleUniversité,IRD,APHM,MEPHI,IHU-MéditerranéeInfection,Marseille,France

b

ServicedeRhumatologie,Hotel-Dieu,CHUNantes,Nantes,France

c

ServicedeMédecineNucléaire,Hotel-Dieu,CHUNantes,Nantes,France

d_{CentredeMedicineNucléaire,AssistancePubliquedesHôpitauxdeMarseille,Marseille,France}

ARTICLE INFO Articlehistory: Received9April2018

Receivedinrevisedform23May2018 Accepted29May2018

CorrespondingEditor:EskildPetersen, Aar-hus,Denmark Keywords: Qfever Coxiellaburnetii Acromio-clavicularinfection Qfeverserology ABSTRACT

Coxiellaburnetiiacromioclavicularinfectionisanewinfectiousfocus,evidencedhereforthefirsttime usingthegoldstandard,culture.Positronemissiontomographyhadacrucialroleinidentifyingthedeep infectiousfocus,evenwhenC.burnetiiserologicaltitreswerelow.

©2018TheAuthors.PublishedbyElsevierLtdonbehalfofInternationalSocietyforInfectiousDiseases. ThisisanopenaccessarticleundertheCCBY-NC-NDlicense( http://creativecommons.org/licenses/by-nc-nd/4.0/).

InJuly2017, a64-year-oldwomanpresented tothehospital withjointpainintheleftshoulderandrightbuttock,whichhad evolved overthe courseof 1week, withoutfever. Hermedical history includedarterial hypertensionand chroniclymphocytic leukaemia,forwhichshehadreceivedallografttreatment2years previously.Shewasstillreceivingprednisoloneorally,20mgper day, and nilotinib, 300mg twice a day, for cutaneous and gastrointestinal graft rejection. On physical examination, she hadweaknessanddecreasedrangeofmotionoftherightshoulder. Shecomplainedofpainintheleftbuttockradiatingtothehindside of the thigh. Laboratory tests showed haemoglobin of 10g/dl, raisedC-reactive protein (94mg/l), and elevated transaminases (alanineaminotransferaseandaspartateaminotransferase45IU/l and 52 UI/L respectively). An ultrasound of the right shoulder revealedacromioclavicularbursitisandmultipleboneerosions.A positronemission tomography(PET) scan identifiedtwo hyper-metabolicfoci,oneintheacromioclavicularjointandoneinthe lefthip(Figure1A ).TheresultsofserologicaltestsforCoxiella burnetiiwerepositive,withphaseIIgGof100,IgMof0,andIgAof 0,andphaseIIIgGof200,IgMof0,andIgAof0.Synovialandbone biopsiesfrombothjointswerethensampled.C.burnetiiPCR(Eldin

et al., 2016a) was positive for both samples, and culture of C. burnetiigrewafter2weeks(Figure1B).Antibioticswerestartedin theformof200mgoforaldoxycyclineonceperdayand200mgof hydroxychloroquinethreetimesperdayfor18months.Atherlast follow-upappointmentinJanuary2018,thepatientshowedsigns ofclinicalimprovement.Thefeverandjointpainhaddisappeared andthepatienthadregainedtherangeofmotionofhershoulder andhipjoints.Theinflammatorysyndromeregressedandhepatic cytolysisimproved.NocontrolPETscanorultrasoundimagingwas performed.

Qfeverisaworldwidezoonosiscausedbytheintracellular bacteriumC.burnetii.Osteoarticularinfectionrepresents1%ofC. burnetii clinical manifestations. This report presents the third caseofanacromioclavicularinfectioncausedbyC.burnetii(Eldin etal.,2016a,b;Angelakisetal.,2016).Evidenceofthebacterium was proved here, for the first time, using three different techniques, including serology, PCR, and culture. The infective focus was identified using two imaging tools. Staphylococcus aureus is the most common microorganism isolated from acromioclavicular joint aspiration. Streptococcus viridans, Streptococcus bovis, and Mycobacterium avium have also been described. Most reports concern patients with an underlying predispositionorsufferingfromanimmunocompromisingdisease (Iyengaretal.,2009).

Although immunosuppression has been considered as a condition predisposingto C. burnetiiinfection, thedata in this

* Correspondingauthorat:IHU,MéditerranéeInfection,19-21BoulevardJean Moulin,13385MarseilleCedex05,France.

E-mailaddress:didier.raoult@gmail.com(D.Raoult).

https://doi.org/10.1016/j.ijid.2018.05.018

1201-9712/©2018TheAuthors.PublishedbyElsevierLtdonbehalfofInternationalSocietyforInfectiousDiseases.ThisisanopenaccessarticleundertheCCBY-NC-ND license(http://creativecommons.org/licenses/by-nc-nd/4.0/).

InternationalJournalofInfectiousDiseases73(2018)27–29

ContentslistsavailableatScienceDirect

International

Journal

of

Infectious

Diseases

regardremaincontroversial,asreportedinrecentstudiesfromthe NetherlandsandFrenchGuiana(Raoult, 1990;Epelboinetal.,2012; Schoffelenetal.,2014).Bycontrast,thecapacityforC.burnetiito evadetheimmuneresponseiswellestablishedanddepends on hostfactors(Raoult,1990;Melenotteetal.,2016).Inarecentlarge prospectivecohortstudy,itwasdemonstratedthat,unlikethecase presentedhere,immunocompromisedpatientswerenotatriskof Qfevercomplications(unpublisheddata). Nonetheless,because the patient was receiving immunosuppressive therapy, the primary infection may have been masked. Moreover, tyrosine kinaseinhibitortherapyhasrecentlybeendescribedasresponsible forchangesinhumoralimmunityoftreatedpatientsinwhomlow levelsofimmunoglobulinandrarecasesofsevere hypogamma-globulinemiahavebeenreported(Rajalaetal.,2017).Thiscould explainthelowserologicaltitreobservedinthepatientdescribed here, who presented a persistent active acromioclavicular C. burnetiiinfection.

PET scanninghas revolutionized the diagnosis of infectious diseasesbyidentifyingfocithatwouldhavegoneunnoticedusing standard computed tomography (Eldin et al., 2016b; Kouijzer etal.,2018).Asillustratedhere,C.burnetiiserologicaltitreswere low.RegardingthecriteriadefinedintheNetherlands,thiscaseof persistentfocalizedC.burnetiicouldhavebeenwronglyexcluded fromthe‘chronicinfection’category(Kampschreuretal.,2015).In immunocompromised patients with fever, chills may not be apparent.In conclusion, serologyis not sufficient todefinethe typeofinfectionrelatedtoQfever.Thesearchforadeepinfectious focus must be systematic, and PET scanning is critical in this approach.

Acknowledgments

WethankNathalieDuclos,EmelineVial,andClioGrimaldier for the culture of the Coxiella burnetii specimen in the NSB3 routine laboratory and the photography performed. This work was supported by the French Government under the “Investissementsd’avenir”(InvestmentsfortheFuture)program, managed by the Agence Nationale de la Recherche (ANR, fr: NationalAgencyforResearch),(reference:MéditerranéeInfection 10-IAHU-03).

Conflictofinterest

Theauthorshavenofinancialconflictsofinterest. References

Angelakis E, Thiberville S-D, Million M, Raoult D. Sternoclavicular joint infectioncausedbyCoxiellaburnetii:acasereport.JMedCaseRep2016;31 (10):139.

EldinC,MelenotteC,MediannikovO,GhigoE,MillionM,EdouardS,etal.FromQ fevertoC.burnetiiinfection:achangeofparadigm.ClinMicrobiolRev2016a;30 (1):115–90.

EldinC,MelenotteC,MillionM,CammilleriS,SottoA,ElsendoornA,etal.18F-FDG PET/CTasacentraltoolintheshiftfromchronicQfevertoCoxiellaburnetii persistentfocalizedinfection:aconsecutivecaseseries.Medicine2016b;95: e4287.

EpelboinL,ChesnaisC,BoulléC,DrogoulAS,RaoultD,DjossouF,etal.Qfever pneumoniainFrenchGuiana:prevalence,riskfactors,andprognosticscore.Clin InfectDis2012;55(1):67–74.

IyengarKP,GudenaR,ChitgopkarSD,RalteP,HughesP,NadkarniJB,etal.Primary septicarthritis of theacromio-clavicular joint:casereportand review of literature.ArchOrthopTraumaSurg2009;129(January(1)):83–6.

Figure1.PETscanimagingandculturefromthejointfluid.(A)WholebodyPETscanimagingshowedhyper-metabolismoftheacromioclavicularjointandthehipjoint.(B) CultureoftheacromioclavicularsynovialbiopsywaspositiveforCoxiellaburnetiiafter2weeks.Culturewasperformedusingtheshell-vialmethodonhumanembryoniclung (HEL)cellswithGimenezstaining.L929cellsarecolouredmalachitegreenandC.burnetiibacteriaarecolouredpinkwithfuchsin.C.burnetiiisanintracellularandfastidious bacteriumlocatedintheintracytoplasmicvacuoles(arrowhead).

KampschreurLM,Wegdam-BlansMC,WeverPC,RendersNH,DelsingCF,SprongT, etal.ChronicQfeverdiagnosis—consensusguidelineversusexpertopinion. EmergInfectDis2015;21(7):1183–8.

KouijzerIJE,KampschreurLM,WeverPC,HoekstraC,vanKasterenMEE,de Jager-Leclercq MGL, et al. The value of 18F-FDG PET/CT in diagnosis and duringfollow-upin273patientswithchronicQfever.JNuclMed2018;59 (1):127–33.

MelenotteC,MillionM,AudolyG,GorseA,DutroncH,RolandG,etal.B-cell non-HodgkinlymphomalinkedtoCoxiellaburnetii.Blood2016;127(1):113–21.

RajalaHLM,MissiryME,RuusilaA,KoskenvesaP,BrümmendorfTH,GjertsenBT, etal.Tyrosinekinaseinhibitortherapy-inducedchangesinhumoralimmunity inpatientswithchronicmyeloidleukemia.JCancerResClinOncol2017;143 (8):1543–54.

RaoultD.HostfactorsintheseverityofQfever.AnnNYAcadSci1990;590:33–8.

SchoffelenT,KampschreurLM,vanRoedenSE,WeverPC,denBroederAA, Nabuurs-FranssenMH,etal.CoxiellaburnetiiCoxiellaburnetiiinfection(Qfever)in rheumatoidarthritispatientswithandwithoutanti-TNFαtherapy.AnnRheum Dis2014;73(7):1436–8.