Splenic abscess due to brucellosis: A case report and a review of the literature

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Case

Report

Splenic

abscess

due

to

brucellosis:

a

case

report

and

a

review

of

the

literature

Mesut

Yilmaz

a,

*,

Ferhat

Arslan

a

,

O

¨ zdil

Bas¸

kan

b

,

Ali

Mert

a

InfectiousDiseasesandClinicalMicrobiologyDepartment,IstanbulMedipolUniversity,SchoolofMedicine,Unkapanı,Atatu¨rkBulvarıNo.27,34083Fatih Istanbul,Turkey

b

RadiologyDepartment,IstanbulMedipolUniversity,SchoolofMedicine,Istanbul,Turkey

1. Introduction

Brucellosisisasystemiczoonoticinfectiousdiseasecausedby Gram-negativebacilliofthegenusBrucella.Althoughthediseaseis prevalentworldwide,itis particularlyendemicinmany Middle Eastern countries, the Mediterranean region, and the Arabian Peninsula.1 _It _occurs _mainly _because _of _the _consumption _of

unpasteurizedmilk and contact withinfected animals. Clinical manifestationsvaryfrommultisysteminvolvementto asymptom-aticinfection.2Almosteveryorganinthebodymaybeinvolved.3 Isolatedsplenicabscessisaveryrareconditionwithaveryhigh mortalityunlesstreatedadequately.4

2. Casereport

A45-year-oldwomanwasadmittedwitha2-monthhistoryof feverwithchills, generalizedmalaise,nightsweats, andweight loss(9kgin2months).Shehadundergoneadilationandcurettage procedureforaﬁrst-trimesterabortion45dayspriortoadmission andhadbeenevaluatedsincethenbyvariousphysiciansforafever

ofunknownorigin(FUO).Shehadnohistoryofrecenttravelor contactwithsickpersonswithasimilarpresentation,butreported theconsumptionofunpasteurizedmilkproducts.

Onphysicalexaminationshewasleanwitharegularperipheral pulseof100bpmandabloodpressureof110/70mmHg.Shewas febrilewithabodytemperatureof39.88C.Herconjunctivaewere pale,buttherewasnoscleralicterusorlymphadenopathy.Cardiac and pulmonaryﬁndings werenormal, and hepatosplenomegaly wasnotdetected.

Laboratory tests revealed the following results: hemoglobin 10g/dl,leukocytecount2.6109_/l,_platelet_count₉₀

109_/l, C-reactive protein 82mg/l, and erythrocyte sedimentation rate 100mm/h.Biochemicalworkup revealednormal serum transa-minasesandkidneyfunctiontests.ChestX-rayand echocardiog-raphywerenormal.AsshewasadmittedwithFUO,brucellosiswas suspected;Brucellaslideandtubeagglutinationtests(Wright,at1/ 640) werepositive. Abdominal computedtomography revealed splenomegalyandmultifocalhypodenselesionsofvarioussizes, withthelargestdiameterof1.5cminthespleen.

Two sets of blood cultures yielded Brucella melitensis. The patient wasthoughtto have multiplesplenic abscesses due to brucellosisandwasstartedonoraldoxycycline(200mg/day)and rifampin(600mg/day)for 6 weeks.Defervescence of the fever occurredonday3andshewasdischargedonday5withoutpatient follow-up and repeat ultrasound appointments. Her clinical conditionimprovedgraduallyandallthelesionshaddisappeared bytheendoftreatment on follow-upultrasoundexaminations. InternationalJournalofInfectiousDiseases20(2014)68–70

ARTICLE INFO Articlehistory:

Received3October2013

Receivedinrevisedform11November2013 Accepted21November2013

CorrespondingEditor:EskildPetersen, Aarhus,Denmark Keywords: Brucellosis Abdominalabscess Splenicdiseases SUMMARY

Splenicabscessduetoacutebrucellosisisarareevent.Wereportacaseofmultiplesplenicabscesses causedbyBrucellamelitensisina45-year-oldwomanandreviewtheEnglishlanguageliteraturebased onaPubMed/MEDLINEsearchofthelast50years.Themajorityofthecasespublishedintheliterature wereduetoB.melitensisandasplenectomywasrequiredinhalfofthecases.Antibioticsalonewithout surgicalinterventioncanbesuccessfulinthetreatmentofpatientswithsplenicbrucellosisintheearly stagesofthedisease.

ß2013TheAuthors.PublishedbyElsevierLtdonbehalfofInternationalSocietyforInfectious Diseases.

* Correspondingauthor.Tel.:+902124607796;fax:+902125317555. E-mailaddress:myilmaz@medipol.edu.tr(M.Yilmaz).

ContentslistsavailableatScienceDirect

International

Journal

of

Infectious

Diseases

j o urn a l hom e pa ge : ww w. e l s e v i e r. c om/ l o ca t e / i j i d

1201-9712 ß2013TheAuthors.PublishedbyElsevierLtdonbehalfofInternationalSocietyforInfectiousDiseases.

http://dx.doi.org/10.1016/j.ijid.2013.11.010

Open access under CC BY-NC-ND license.

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Table 1

Clinical characteristics and outcome of the patients Case No. Author and

reference

Year published

Patient age, years/sex

Risk factor Localization Brucella

antibody titer

Isolate Therapy Outcome

1 Spink14

1964 51/M Farmer Liver/spleen, bone 1:320 B. suis Surgical drainage + TET Recovered

2 Spink14

1964 58/M Butcher Spleen 1:320 B. suis Splenectomy + TET 3 months Recovered

3 Spink14

1964 54/M Cattle buyer Liver/spleen,

lymph node

1:80 B. suis NA Died of hemorrhage from

esophageal varices

4 Spink14 ₁₉₆₄ _53/M _Farmer _Spleen _1:1280 _{B. suis} _{Splenectomy + TET} _Recovered

5 Spink14

1964 53/M Farmer Spleen 1:1280 None Splenectomy + antibiotic (NA) Recovered

6 Ates et al.15

1992 30/F NA Liver/spleen 1:1280 B. melitensis TET + STREP IM 4 weeks Recovered

7 Ates et al.15

1992 50/M NA Spleen 1:1280 B. melitensis TET + STREP IM 4 weeks then

RIF 4 weeks

Recovered

8 Saadeh et al.18 ₁₉₉₆ _23/M _Farmer _{Spleen, aortic valve} _1:2560 _{B. melitensis} _{RIF + DOX 6 months,}

splenectomy

Recovered

9 Solera et al.19

1996 49/F None Spleen 1:640 None Antibiotics (NA), splenectomy Recovered

10 Colmenero et al.11

2002 53/M History of brucellosis Spleen 1:80 Serum B. melitensis

DNA by PCR

2 cycles of DOX 2 months + STREP IM 21 days

Relapsed

2002 80/NA History of brucellosis Spleen 1:320 Tissue B. melitensis

DNA by PCR

DOX 2 months and STREP IM 21 days + splenectomy followed by (DOX + RIF) 3 months

Died of other causes

2002 72/NA History of brucellosis Spleen 1:40 Serum and tissue B.

melitensis DNA by PCR

DOX 2 months and STREP IM 21 days + splenectomy followed by (DOX + RIF) 3 months

Recovered

13 Yayli et al.20

2002 70/F Unpasteurized milk

consumption

Spleen 1:320 B. melitensis (DOX + RIF) 6 weeks and

STREP IM 21 days

Recovered

14 Yilmaz et al.21 ₂₀₀₃ _19/M _{Unpasteurized milk}

consumption

Spleen, aortic valve High titer B. melitensis Splenectomy + antibiotic (NA) Recovered 15 Ruiz Carazo et al.22

2005 60/M NA Spleen NA NA Splenectomy + antibiotic (NA) Recovered

16 Del Arco et al.16

2006 39/F Unpasteurized milk

consumption

Spleen 1:1280 B. melitensis

DNA by PCR

DOX 6 weeks + STREP IM 2 weeks, splenectomy, RIF + DOX 1 month

Recovered

17 Sayilir et al.10 ₂₀₀₈ _61/M _NA _Spleen _1:1280 _Negative _{RIF + DOX and TMP–SMZ} _Recovered

18 Park et al.17

2009 45/M Livestock industry worker Spleen, aortic valve 1:160 B. abortus GEN and RIF + DOX + TMP–SMZ

12 months

Recovered

19 Eruz et al.9

2011 52/F Raw meatball consumption Spleen, respiratory

system

1:640 B. melitensis RIF + DOX 6 weeks Recovered

20 Deveer et al.8

2013 21/M Farmer Spleen 1:1280 B. melitensis (DOX + CIP) 12 weeks + STREP

IM 3 weeks

Recovered

21 Present case 2013 45/F Unpasteurized milk

consumption

Spleen 1:640 B. melitensis (RIF + DOX) 6 weeks Recovered

M, male; F, female; TET, tetracycline; DOX, doxycycline; STREP, streptomycin; GEN, gentamicin; RIF, rifampin; CIP, ciproﬂoxacin; TMP–SMZ, trimethoprim–sulfamethoxazole; NA, not available; IM, intramuscular.

M. Yilmaz et al. / International Journal of Infectious Diseases 20 (2014) 68–70 69

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The patient remained asymptomatic without any signs or symptomsof diseaserecurrenceat 1 yearafterthecompletion oftreatment.

3. Discussion

Sinceourpatienthadahistoryofconsumptionofunpasteurized milk products, the mode of transmission was thought to be ingestion of contaminated food. As a multisystem disease, brucellosiscanoftenbea diagnosticdilemmaas thediseaseis associatedwithawidevarietyofsignsandsymptoms.Ourpatient hadadurationofsymptomsof2monthsonpresentation,whichis typicalofFUO.

Based on the initiation of her symptoms, our patient had brucellosisduringherpregnancy.Thereisevidencethat brucello-siscaninduceabortioninhumans.Positivecultures ofBrucella from human placenta, aborted fetuses, and other products of conceptionhavebeenreportedpreviously.5Moreover,replication ofB.melitensisinhumantrophoblastshasrecentlybeenshown.6

Therefore,itisspeculatedthatbrucellosiscausesfewer spontane-ous abortions in humans than animals due to the absence of erythritolinthehumanplacentaand fetus.5 _Our _patient_might

havehadtheabortionduetobrucellosis.

Focal forms of brucellosis may occur in up to a third of patients.3,7 _As _they _are _considered _true _{complications} _of _the

disease,theytendtohaveaworseprognosiscomparedtothe non-focal forms. Osteoarticular, neurological, genito-urinary, liver, hematological,and cardiacinvolvementsare themostfrequent focal forms. Isolated splenic abscess is an extremely rare and seriouscomplicationofbrucellosis.8–10_Not_a_single_case_was_found

inoneofthelargestTurkishseriesof1028cases.3Nevertheless,its incidencehasbeenreportedasless than2%inotherlarge case series.11,12 _Spontaneous _splenic _rupture _due _to _brucellosis _has

beenreportedinafewrarecases.13

Our PubMed/MEDLINE search (1964–2013) yielded20 adult casespublishedintheEnglishlanguageliteraturetodate.Baseline demographicsandclinicalcharacteristicsofallthecasesaregiven inTable1.8–11,14–22_The _mean_age_of_the_patients_was₄₉ _years

(range19–80years)and68%weremen.Fourofthecasesweredue toBrucella suis and only one was due to Brucella abortus; the remaining70%wereduetoB.melitensis.Noneofthepatientsdied duetosplenicabscessoranyothercomplicationofbrucellosis.A splenectomywasrequiredin10(47%)cases.Insomecaseswith chronicsplenic abscesswhereserumagglutinationremainedas lowas 1:40,diagnosis wasestablishedusing PCRof peripheral bloodandsplenictissue.11

The best therapeutic approach for splenic abscess due to Brucellahasnotbeenclearlyestablished.Thetreatmentofsplenic abscesswithantibiotics alone hasbeenreported, however this modalityappearstobesuccessfulonlyintheearlystagesofthe disease when there is no calciﬁcation in the lesions.4,15,20 _A

therapeuticapproachintheearlystages,withtheuseofantibiotic therapyaloneorassociatedwithnon-surgicaldrainage,maybean initialoption,butprolongedtherapyoverseveralmonthsmaybe requiredandcarefulfollow-upisessentialbecausethecomplete cureofthediseasecannotbeguaranteed.12,16_The_treatment _of

chronic lesions should involve a combination of medical and surgicaltherapy.16 _Our _patient _responded _well_to_the _standard

duration6weeksoftreatment.

Inconclusion,splenicabscessduetobrucellosisisarareentity and prompt diagnosis is crucial for initiating therapy and

preventingcomplications.Inendemicareas,patientswithacute brucellosisshouldbeinvestigatedforsplenicabscessby abdomi-nalultrasonography,particularlywhentheyhavehadaprolonged fever.Theresponsetoantibiotictherapyaloneisfavorableinthe early stages, however careful follow-up is required to ensure completecureofthedisease.

Conﬂictofinterest:Theauthorsreportnoconﬂictsofinterest. Theauthorsaloneareresponsibleforthecontentandwritingofthe paper.Nosupportregardingeditorialassistanceand/or prepara-tionofthearticlewasreceived. Duringthestudynofundingor support of any kind was received from any organization or company.

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