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 EMHJ • Vol. 16 No. 4 • 2 010  Eastern Mediterranean Health Journal La Revue de Santé de la Méditerranée orientale 448 Case report Paravertebral abscess and neurological decits in cervical brucellar spondylitis V. German, 1  N. Papadopoulos, 1  C. Diakalis, 1  C. Goritsas 1  and A. Ferti 1 1  Sotiria General Hospital o f Athens, Department of Interna l Medicine, A thens, Greec e (Corres pondence to N. Papad opoulos : npnck7@ya hoo.com). Received: 30/01/08; accepted: 03/04/08 Introduction Brucellosis is a zoonotic infection with a worldwide distribution, endemic in the Mediterranean region, and is as - sociated with high morbidity in humans [1,2]. Osteoarticular complications are the most common focal complications of  Br uce lla  spp. infection [ 3]. ese complications commonly a ect the axial skeleton, with vertebral spondylitis accounting for 35%–50% of all osteoar - ticular complications [4–6 ]. Estimates of the incidence of spondylitis range from 9% to 31% [ 4,5 ]. While lumbar spine involvement is the most common, cervical involvement is a rare, but more severe, complication, accounting for 8.3% of cases of vertebral osteomyelitis in a recent study [4].  We pr es en t a ca se of br uc el la r spondylitis with paravertebral mass in the cervical segment that resulted in neurological decits. Case report  A 36 -ye ar- old fe ma le pa tie nt , a ve t - erinarian who was working and living in Albania, with a history of neph rolithi - asis and an unremarkable family history, presented at our hospital with high fever that had started 20 days previously. Co - existing symptoms were night sweats, headache, neck pain extending down the back, and myalgias, especially in the right arm. e patient had initially been treated by a general practitioner with ciprooxacin for 1 week, followed by ampicillin and streptomycin for 4 days  wit h app are ntl y no rem iss ion of th e symptoms. e Wright test was report - ed as negative at that time. e patient presented at the Sotiria General Hos - pital for Chest Diseases in Athens with persistent high fever (up to 39.8 C), neck and back pain, myalgias and re - stricted movement of the right arm. Physical examination revealed nothing but cervical spine tenderness at C4–C6, while neurological examina - tion revealed a decrease in strength in the right biceps and triceps, with aenu- ation of their reexes. Laboratory results showed an eryth - rocyte sedimentation rate of 99 mm/h, a leukocyte count of 9480 /mm 3  with 49% neutrophils and 32% lymphocytes, a haemoglobin level of 11.8 g/dL and a platelet count of 348 000 /mm 3 . Liver function tests were abnormal: alkaline phosphatase 1484 U/L, gamma- glutamyl-transpeptidase 443 U/L, serum glutamic oxaloacetic transami - nase 134 U/L, serum glutamic pyruvic transaminase 324 U/L and lactate dehydrogenase 348 U/L. Blood urea, creatinine, total bilirubin and electrolyte levels were normal. Four consecutive  blood cultures were negative. ere was a di use hypergammaglobulinaemia and an increase in C-reactive protein  value to 3.41 mg/L (normal value < 0.8 mg/L). Tests for rheumatoid factor and antinuclear antibodies, the tuberculin skin test and Wright agglutination test  were all negative.  Bru cel la  infection was conrmed  with a positi ve result for  Brucel la IgM antibodies (> 100 U/mL) using an enzyme-linked immunsorbent assay (Serion Immundiagnostica GmbH, Germany). Cervical spine X-ray showed nar - rowed C5–C6 and C6–C7 intraverte -  bral spaces and a possible paravertebral anterior mass in the C6–C7 area. o - racic spine X-ray had no abnormal ndings. A cervical spine computerized tomography (CT) scan consequently performed revealed spondylodiscitis in the C5–C6 intravertebral space, with destructive changes in the C5 vertebral  body and presence of tissue in the right lateral paravertebral space. Cervical magnetic resonance imaging (MRI) conrmed the C5–C6 spondylitis and the existence of a minimal inammatory paravertebral mass with root compres - sion (Figure 1). e patient underwent a CT guided ne-needle aspiration of the abscess. Histological examination revealed a nonspeci c inammatory inltration with no evidence of malig - nancy. All culture tests were negative. e patient was started on oral doxy- cycline 100 mg 2 × day, oral rifampicin 600 mg daily and intramuscular strep - tomycin 1 g 4 × day. e doxycycline and rifampicin regimen continued for a period of 3 months and streptomycin  was discontinued aer 2 weeks. irty days aer admission, the patient was

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  • EMHJ Vol.16 No.4 2010 EasternMediterraneanHealthJournalLaRevuedeSantdelaMditerraneorientale

    448

    Case report

    Paravertebral abscess and neurological deficits in cervical brucellar spondylitisV. German,1 N. Papadopoulos,1 C. Diakalis,1 C. Goritsas1 and A. Ferti1

    1Sotiria General Hospital of Athens, Department of Internal Medicine, Athens, Greece (Correspondence to N. Papadopoulos: [email protected]).

    Received: 30/01/08; accepted: 03/04/08

    Introduction

    Brucellosis isazoonotic infectionwithaworldwidedistribution, endemic intheMediterranean region, and is as-sociatedwithhighmorbidityinhumans[1,2].OsteoarticularcomplicationsarethemostcommonfocalcomplicationsofBrucella spp. infection [3].Thesecomplications commonly affect theaxialskeleton,withvertebralspondylitisaccountingfor35%50%ofallosteoar-ticularcomplications[46].Estimatesof the incidenceof spondylitis rangefrom9% to31%[4,5].While lumbarspineinvolvementisthemostcommon,cervicalinvolvementisarare,butmoresevere, complication, accounting for8.3%ofcasesofvertebralosteomyelitisinarecentstudy[4].

    We present a case of brucellarspondylitiswithparavertebralmass inthe cervical segment that resulted inneurologicaldeficits.

    Case report

    A 36-year-old female patient, a vet-erinarianwhowasworkingand livinginAlbania,withahistoryofnephrolithi-asisandanunremarkablefamilyhistory,presentedatourhospitalwithhighfeverthathadstarted20dayspreviously.Co-existing symptomswerenight sweats,headache,neckpain extendingdowntheback,andmyalgias,especiallyintherightarm.

    The patient had initially beentreatedby a generalpractitionerwithciprofloxacin for1week, followedbyampicillinandstreptomycin for4dayswith apparently no remission of thesymptoms.TheWrighttestwasreport-edasnegativeat thattime.Thepatientpresentedat theSotiriaGeneralHos-pitalforChestDiseasesinAthenswithpersistenthigh fever (up to39.8 C),neck andbackpain,myalgias and re-strictedmovementoftherightarm.

    Physical examination revealednothingbut cervical spine tendernessatC4C6,whileneurologicalexamina-tion revealedadecrease in strength intherightbicepsandtriceps,withattenu-ationoftheirreflexes.

    Laboratoryresultsshowedaneryth-rocytesedimentationrateof99mm/h,a leukocytecountof9480/mm3with49%neutrophilsand32%lymphocytes,ahaemoglobinlevelof11.8g/dLandaplateletcountof348000/mm3.Liverfunction testswereabnormal:alkalinephosphatase 1484 U/L, gamma-glutamyl-transpeptidase 443 U/L,serumglutamicoxaloacetic transami-nase134U/L,serumglutamicpyruvictransaminase 324 U/L and lactatedehydrogenase348U/L.Bloodurea,creatinine,totalbilirubinandelectrolytelevelswerenormal.Four consecutivebloodcultureswerenegative.Therewasa diffuse hypergammaglobulinaemiaandan increase inC-reactiveproteinvalueto3.41mg/L(normalvalue100 U/mL) using anenzyme-linked immunsorbent assay(Serion Immundiagnostica GmbH,Germany).

    Cervical spineX-ray showednar-rowedC5C6andC6C7 intraverte-bralspacesandapossibleparavertebralanteriormassintheC6C7area.Tho-racic spine X-ray had no abnormalfindings.Acervicalspinecomputerizedtomography(CT)scanconsequentlyperformedrevealedspondylodiscitis intheC5C6 intravertebral space,withdestructivechangesintheC5vertebralbodyandpresenceoftissueintherightlateral paravertebral space. Cervicalmagnetic resonance imaging (MRI)confirmed theC5C6spondylitisandtheexistenceofaminimalinflammatoryparavertebralmasswith rootcompres-sion(Figure1).ThepatientunderwentaCTguidedfine-needleaspirationoftheabscess.Histological examinationrevealed a nonspecific inflammatoryinfiltrationwithnoevidenceofmalig-nancy.Allculturetestswerenegative.

    Thepatientwasstartedonoraldoxy-cycline100mg2day,oral rifampicin600mgdailyand intramuscular strep-tomycin1g4day.Thedoxycyclineand rifampicin regimencontinued foraperiodof3monthsandstreptomycinwasdiscontinuedafter2weeks.Thirtydays after admission, thepatientwas

  • 449

    dischargedfreeofsymptomswithamel-iorationof theneurological signs.Thelaboratory testsonherdischargewerenormal,includingerythrocytesedimen-tationrateandliverfunctiontests.

    Threemonths after discharge thepatientremainedfreeofsymptoms,neu-rologicalexaminationwasnormalandcomplete laboratory testswerewithinnormallimitswithadecreaseintheanti-BrucellaIgMantibodytitreto50IU/mL.

    AsecondcervicalMRIshowedremis-sionofspondylitiswithnoevidenceof

    theparavertebralmass(Figure2).

    Discussion

    Brucellosisremainsanimportantpublic

    healthproblem in theMediterranean

    region,whereefforts toeradicateBru-cella melitensis inanimalshave failed. In

    Greece,284casesofbrucellosiswereofficiallyreportedduring2006[7].

    Theclinicalmanifestationsofbru-cellosisarevaried.Themusculoskeletalsystem is frequently affected and thespineisthemostcommonsiteofbonebrucellosis.Thespineismostfrequentlyaffectedinthelumbosacralregion,andpatientsareusuallymalesovertheageof50years[1,4,5].Cervical involvement,however, is accompaniedbymore se-veremanifestations, suchasparaverte-bralandepiduralmasses,neurologicalandfunctionaldisabilities[1,5].

    Diagnosisandtreatmentshouldbequickand intense, sincedelay in treat-ment leadstoahighmorbidityrate. Inamulticentreprospectivestudyof593patientswithbrucellosis. the incidenceof spondylitiswas9.7%[5].The inci-denceofmedullaorrootcompression,however,dependedontheaffectedseg-mentofthespinalcolumn.Inthegroupof patientswith cervical spondylitis,71%hadcompressionof themedullaor roots, versus 11% and21% in thedorsal and lumbargroup respectively.Inaddition,patientswithcervical anddorsal spondylitis had a significantlyhigher rateofparavertebralorepiduralmassesthanthoseinthelumbargroup.Moreover, therewere a significantlyhighernumberofpatientswithunsat-isfactoryprogress in thecervicalgroupcomparedwith thedorsaland lumbargroups.RecentstudiesinGreecefoundosteoarticular involvement in nearlyhalfthecasesofacutebrucellosis[2,8].

    Occasionallypatientswithparaver-tebral abscess fail to respond to con-servativetreatmentandhavetoundergosurgery[1,9];however,successhasbeenreportedwithconservative treatment,andthismaybesufficientinselectedpa-tients [10].Generally, little informationisavailableaboutbrucellar spondylitisandtreatmentregimensvarywidely.TheIoanninarecommendations,developedbyexperts inNovember2006,advisedthattheoutcomeofspondylitismaybeimprovedwhenastreptomycinregimen

    Figure 1 Pre-treatment case of brucellar spondylitis. (A) Sagittal T1W image: spondylitis is depicted as low signal intensity lesion of the C5 and C6 vertebral bodies. (B) Transverse FFET2W image: the paravertebral inflammatory mass is indicated as high-signal intensity lesion protruding mainly to the left intervertebral foramen, compressing the homolateral nerve root

    Figure 2 Post-treatment case of brucellar spondylitis. (A) Sagittal T1W image: resolution of the spondylitis is demonstrated; C5 and C6 vertebral bodies have normal signal intensity. (B) Transverse FFET2W image: no paravertebral inflammatory mass is found post-treatment and no root compression is depicted

  • EMHJ Vol.16 No.4 2010 EasternMediterraneanHealthJournalLaRevuedeSantdelaMditerraneorientale

    450

    Solera J et al. Brucellar spondylitis: review of 35 cases and litera-1. ture survey. Clinical infectious diseases, 1999, 29:14409.

    Pappas G et al. Brucellosis. 2. New England journal of medicine, 2005, 352(22):232536.

    Colmenero JD et al. Complications associated with 3. Bru-cella melitensis infection: a study of 530 cases. Medicine, 1996, 75:195211.

    Colmenero JD et al. Clinical findings, therapeutic approach, 4. and outcome of brucellar vertebral osteomyelitis. Clinical infec-tious diseases, 2008, 46:42633.

    Colmenero JD et al. Clinical course and prognosis of brucella 5. spondylitis. Infection, 1992, 20:3842.

    Basaranoglu M et al. A case of cervical brucella spondylitis with 6. paravertebral abscess and neurological deficits. Scandinavian journal of infectious diseases, 1999, 31:2145.

    Epidemiologic data for infectious diseases. 7. Athens, Hellenic Cen-tre for Infectious Diseases Control, Department of Epidemio-

    References

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    Andriopoulos P et al. Acute brucellosis: presentation, diagno-8. sis, and treatment of 144 cases. International journal of infectious diseases, 2007, 11:527.

    Tekkok IH et al. Brucellosis of the spine. 9. Neurosurgery, 1993, 33:83844.

    Guzey FK et al. Cervical spinal brucellosis causing epidural and 10. prevertebral abscesses and spinal cord compression: a case report. Spine journal, 2007, 7:2404.

    Ariza J et al. Perspectives for the treatment of brucellosis in the 11. 21st century: the Ioannina recommendations. Public Library of Science medicine, 2007, 4(12):18728.

    Malavolta N et al. Brucella spondylitis with paravertebral ab-12. scess due to Brucella melitensis infection: a case report. Drugs under experimental and clinical research, 2002, 28:958.

    isusedandthatthedurationoftherapy

    mustbenotlessthan3months[11].Inarecentstudyof918patientswith

    brucellosis, the incidence of vertebral

    osteomyelitiswas10.4%,andcervicalin-volvementwasestablishedin8.3%ofthem

    [4]. In that studypatientswere treatedwithdoxycyclinefor3monthsplusstrep-tomycin for2or3weeks(DOX-STR

    regimen)ordoxycyclineplusrifampicin

    (DOX-RIFregimen)bothfor3months.

    No significantdifferenceswere found

    betweenthe2treatmentgroupsintreat-mentfailure,mortalityandrelapse.Inour

    case,wepreferredtouseinitiallystrepto-mycinfor2weeksplusdoxycyclineandrifampicin fora totalof3months.Theadministrationoftripleregimensisusedforneurobrucellosis[2].Webelievethatthistreatmentstrategycombinesthesu-periorityoftheDOX-STRregimenwiththe better implementationof theoralDOX-RIFregimen.However,morewell-designed,controlled trialsareneededtoestablishthesuperiorityofacertainregi-men.Currentlytherapeuticdecisionsareguidedmainlybyexpertopinion.Inourcase therewasa rapidresponse toanti-microbialchemotherapy,aswasreported

    inpreviouscasesofbrucellarspondylitis

    withparavertebralabscess[6,12].Cervical spondylitis should be

    considered a severe complication of

    brucellosis; therefore its treatmentand

    follow-upmustbeenergeticinorderto

    detectandcorrectasearlyaspossible

    compressionsof theneuralaxisand its

    roots.Treatmentshouldbeprolonged

    topreventbothcomplicationsand re-lapses.Furtherevaluation incontrolled

    studies isneeded toassess theefficacy

    andsafetyoftreatmentoptionsinbrucel-larspondylitis.