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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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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.