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CASE REPORT – OPEN ACCESS

InternationalJournalofSurgeryCaseReports69(2020)109–113

ContentslistsavailableatScienceDirect

International Journal of Surgery Case Reports

j o u r n al ho m e p a g e :w w w . c a s e r e p o r t s . c o m

An unusual case of extensive contiguous cervicothoracic spinal tuberculosis involving fourteen damaged segments: A case report

Ifran Saleh

^a,∗

, Didik Librianto

^b

, Phedy Phedy

^b

, Toto Suryo Efar

^c

, Anissa Feby Canintika

^c

aDepartmentofOrthopaedics&Traumatology,CiptoMangunkusumoNationalCentralHospital,JalanDiponegoroNo.71,JakartaPusat,10430,Indonesia

bDepartmentofOrthopaedics&Traumatology,FatmawatiGeneralHospital,JalanRSFatmawatiNo.1,CilandakKotaJakartaSelatan,12430,Indonesia

cDepartmentofOrthopaedics&Traumatology,FacultyofMedicineUniversitasIndonesia,JalanSalembaNo.4,JakartaPusat,10430,Indonesia

a r t i c l e i n f o

Articlehistory:

Received11December2019

Receivedinrevisedform30January2020 Accepted2February2020

Availableonline6February2020

Keywords:

Cervicothoracicspinaltuberculosis Multilevelcontagiousinvolvement

a b s t r a c t

INTRODUCTION:Cervicothoracicspinaltuberculosis(CTSTB)isarareanddisablingdiseaseinvolvingthe mobile,transitionalzonebetweenthelordoticcervicalandthekyphoticthoracicspine.Approximately halfofthosecasesinvolvesoneortwosegmentsofcervicothoracicvertebrae.Wereporteda28-year-old femalewithtuberculousinvolvementoffourteencontiguousvertebralsegments.

PRESENTATIONOFCASE:A28-year-oldfemalepresentedwithtuberculousinvolvementoffourteencon- tiguousvertebralsegmentsispresented.AseriesofradiographicandCTscandepictedmultiplevertebral bodydestructionanteriorly,alongwithfacetjointdislocationandmildretrolisthesisofC4-C5seg- ments.MRimagesofthecervicalregionwasdemonstratedpathologiccontrastenhancementonC4 toT7vertebrae,atotaloffourteencontiguoussegments.

DISCUSSION:Ofallspinaltuberculosis,CTSTBaccountsforonly5%.Inadditiontoitsrarityasasitefor tuberculosis,thecervicothoracicjunctionhasanatomicalandclinicalpeculiarities,asareversalofthe mobile-lordoticcervicalvertebraetorigid-kyphoticthoracicvertebraeoccursatthislocation.MostCTSTB involvesonlytwosegments;however,inthiscase,wefoundaveryextensivecasewhereintherewere fourteendamagedsegments.

CONCLUSIONS:OurreportdemonstratesoneofthelongestinvolvementofextensivecontiguousCTSTB whowastreatedwithone-stageposterior-onlyapproach.However,asthisisonlyareportofonecase, furtherstudiesarerequiredtoinvestigatethesafetyandefﬁcacyofsuchapproachfortreatingextensive CTSTB.

1. Introduction

Cervicothoracicspinaltuberculosis(CTSTB),deﬁnedbytuber- culosisinfectioninvolvingthevertebraebetweenC7toT3,is a rarediseasethattypicallyresultsindisablingcomplicationssuch askyphoticdeformity,largeparavertebralabscesses,andprogres- sivespinal corddamage with severeneurological deﬁcit [1–3].

Thisdiseaseconstitutesonly5%ofallcasesofspinaltuberculosis [4],anditoftenpresentsasatreatmentchallengeforspinesur- geons.Thecervicothoracicjunctionisanatomicallylocatedinthe transitionalzonebetweenthemoremobile,lordoticcervicaland themorerigid,kyphoticthoracicspine[1].Sincethejunctionis weight-bearingstructure,destructionofsuchstructurebytubercu- losisinfectionnotuncommonlyresultsinnumerouscomplications aforementioned above. Moreover, the unique interrelationship ofcervicothoracicvertebraecommandsdifﬁcultexposureofthe

∗Correspondingauthor.

E-mailaddress:totosuryoefar@gmail.com(I.Saleh).

infectionfocus,bothanteriorlyandposteriorly,makingoperative treatmentaconsiderablechallenge[1].Approximatelyhalfofcer- vicalspinaltuberculosisinvolvesatmosttwo segments.In this report,wepresenta caseof28-year-oldfemalediagnosedwith CTSTBwithinvolvementofcontiguousfourteenvertebrae.

2. SCAREcriteriacompliance

ThisworkhasbeenreportedinlinewiththeSCAREcriteria[5].

3. Casereport

A28-year-oldfemalepresentedtoFatmawatiHospital,Jakarta, Indonesia due to progressive tetraparesis accounting for seven months.Initiallytherewasmildneckpain,andthensheexperi- encednumbnessalongwithweaknessofallfourextremities.The symptomsweregraduallyworsenedsuchthatatthetimeofadmis- sion,shecouldnotwalk.

AseriesofradiographicandCTscandepictedmultipleverte- bralbodydestructionanteriorly,alongwithfacetjointdislocation

https://doi.org/10.1016/j.ijscr.2020.02.003

org/licenses/by/4.0/).

Downloaded for Anonymous User (n/a) at University of Indonesia from ClinicalKey.com by Elsevier on May 12, 2022.

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Fig.1.Preoperativeradiographofthepatient.

Fig.2.PreoperativeCTimagesdemonstratedextensivevertebralbodydestruction, severalfacetjointdislocationsandmildretrolisthesisofC4toC5segment.

andmildretrolisthesisofC4-C5segments(Figs.1,2).MRimagesof thecervicalregionwasdemonstratedpathologiccontrastenhance- mentonC4toT7vertebrae,atotaloffourteencontiguoussegments, withspinalcanalstenosisonthelevelofC4toT4andbilateral anterolateralparavertebralsofttissueabscessatthelevelofC4to T9(Fig.3).Thoseﬁndingswerehighlytypicalofspinaltuberculo- sis.

Thepatientwasadministeredanti-tuberculousagentsfortwo months before undergoing one-staged posterior-only debridement,decompression,andinstrumentation.Postoperativeradio- graphsdemonstratedthatthekyphosiswasobviouslyimproved (Fig.4).Afterthesurgery,thepatientwasputona cervicothoracic brace. The patient was closely followed up, and after 12 monthspostoperatively,sheregainedneurologicalrecoverywith onlymildresidualneckpain.Thepatientdemonstrateda satis-

fyinglevel of functionalimprovement asrecorded bythe Neck DisabilityIndex(NDI)andSF-36scoresof4/100and94%,respectively.

Bony bridgeon CT imageswas discovered at 18 months of follow-up,alongwiththenormalvalueoferythrocytesedimen- tationrateandC-reactiveprotein;itwasthetimewediscontinued theanti-tuberculoustherapy.

4. Discussion

Tuberculosis, caused by Mycobacterium tuberculosis, remains oneoftheoldestdiseasesworldwide.Despiteanancientdisease, tuberculosisremainsanimportantproblem,particularlyinunder- developed countries.Extra-pulmonary tuberculosisaccountsfor 15–20%ofalltuberculosiscases[6],andthemostcommonform ofthisspectrumisspinaltuberculosis,whichconstitutesof50%

ofallskeletaltuberculosiscases[7].Spinaltuberculosisremainsa greatchallengetophysiciansdoitsnonspeciﬁcsymptomsthatmay resultindelayofdiagnosis,aswellassigniﬁcanthighmorbidityand mortality.

Ofallspinal tuberculosis,CTSTB accountsfor only5%[4]. In additiontoitsrarityasasitefortuberculosis,thecervicothoracic junctionhasanatomicalandclinicalpeculiarities,asareversalof themobile-lordoticcervicalvertebraetorigid-kyphoticthoracic vertebraeoccursatthislocation[8–10].Infact,ofallregionsofthe spine,thecervicothoracicjunctionisarguablythemostchallenging entity[11].Moreover,suchjunctionpresumesspeciﬁcbiomechan- icsandstabilitydifferentfromotherspinalregions[9,12].Affecting mainlytotheanteriorcolumn,contiguoustuberculouslesionof cervicothoracicjunctionleadstoprofoundinstabilityandaltered biomechanicsoftheweight-bearingarea.CTSTBwasalsoassoci- atedwithhighdegreeofspinalcordcompressionthatinvariably leadstoneurologicaldeﬁcit[8].

MostCTSTBinvolvesonlytwosegments;however,inthiscase, wefoundaveryextensivecasewhereintherewerefourteendam- agedsegments.Inaseriesof20patientswithCTSTB,Lanetal.[13]

foundthat11(55%)ofthepatientshadtwodamagedsegments,

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CASE REPORT – OPEN ACCESS

I.Salehetal./InternationalJournalofSurgeryCaseReports69(2020)109–113 111

Fig.3.SagittalcervicothoracicMRimagesdemonstratedpathologiccontrastenhancementonC4toT7segmentsalongwithvertebralbodyinvolvementandspinalcanal compressionatthelevelofC4toT4.FromcoronalMRimages,alargeparavertebralabscesswaspresentedanteriorly.

Fig.4.Postoperativeradiographdemonstratedpediclerodandscrewconstructthatresultedinimprovedkyphoticdeformity.

andthemostextensivecaseonlyinvolvedthreesegments,which onlyoccurredinone(5%)subject.Inacaseseriesof10patients,6 (60%)subjectshadthreedamagedsegments,andthemostexten- sivecaseinvolved8segments.Todate,thereisnopublishedreport regarding CTSTB involvingmorethan tensegments; this isthe ﬁrstreportthatreportsCTSTBinvolvingfourteendamagedseg- ments.

Atpresent,CTSTBisstillrarelyreported;thus,therehasbeenno speciﬁcconcensus,letalonefortheextensiveone.Indicationsfor surgeryinCTSTBincludekyphosisof≥20^◦,instability,neurologi- calcompromise,andpersistentpain[14].CTSTBcouldbemanaged withanterior-onlydebridementwithorwithoutinstrumentation, andcombinedanteriordebridementfollowedbyposteriorinstru- mentation.Inthiscasereport,weperformedone-stageposterior transpediculardebridement,decompression,andinstrumentation.

Severalauthorsrecommendedtheanteriordebridementwithor withoutinstrumentationforthemaintreatmentofCTSTBdueto

itsaccesstotuberculouslesionanditsparavertebralabscess,which occursanteriorly[1,15,16].However,itisoftenconsidereddifﬁ- cultduetothecomplexstructureofthecervicothoracicjunction.

Manystructures,includingthoracicbones,clavicles,costalbone, largebloodvessels, etc,covertheregion[1,9];thus,theopera- tivefieldisnarrow,whichpresentsasignificantchallengetothe spinesurgeon[17].Recently,theposterior-onlysurgeryhasgar- neredinterestsasanalternativetreatmentforCTSTB,asitissimple, labelledwithgoodclinicalefficacyandfewcomplications[9,18].

Feyzaetal.[19]andRath etal.[20] reportedgoodneurological resultsafterposteriordebridementandinternalﬁxationinthose withneurologicalimpairmentduetoCTSTB.Theresultsweresim- ilartothoseobtainedviaanteriorapproach.Zengetal.[9]found thatposteriorinstrumentationmayprovidebetterkyphoticcorrec- tionandisbeneﬁcialtothestressdispersion,whichcaneffectively preventimplantfailure.Severalpublishedstudiesregarding the managementofCTSTBarepresentedinTable1.

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CASE REPOR T – OPEN A CCESS

I.Salehetal./InternationalJournalofSurgeryCaseReports69(2020)109–113

anteriorandposterior surgery(groupB)and25 underwentone-stage posteriorsurgery(groupC).

improvedkyphosis(p<0.001)

Zhuetal.[4] 2018 China Prospective 45 CervicothoracicTB 29:16 35.4(17–62) 6.6yearson average(range 3–13years)

19patientsweretreated withasingle-stageanterior debridement,fusionand instrumentationapproach, andtheother26patients weretreatedwitha single-stageanterior debridementandfusion, posteriorfusionand instrumentationapproach

ThekyphosisangleandNDI andJOAscoreswere signiﬁcantlychangedfrom preoperativevaluesof 34.7±6.8^◦,39.6±4.6and 10.7±2.8topostoperative valuesof10.2±2.4^◦,11.4±3.6 and17.6±2.4,respectively (p<0.05).Asidefromone recurrentpatient,bonefusion wasachievedintheother44 patientswithin6–9months (mean7.2months)

Mahadewa[22] 2016 Indonesia CaseReport 1 CervicothoracicTB 1:0 12 6months One-stagelaminmectomy

decompressionand stabilizationfusionvia posteriorapproach

Completeresolutionofall neurologicaldeﬁcitsexceptfor verymildgaitidisturbance Zhangetal.[1] 2015 China Prospective 15 CervicothoracicTB

withkyphosis

7:8 40.9(17–67)years 27.7±8.8 one-stagesurgical treatmentbyposterior ﬁxation,anterior debridement,bone grafting,andanterior ﬁxation

Bonefusionwasachieved withinthreetosixmonths (average,5.5months).Inthe 15cases,nopostoperative severecomplicationsoccurred andneurologicfunctionwas improvedinvariousdegrees.

Lanetal.[13] 2011 China Prospective 20 CervicothoracicTB 17:3 N/A 16–39months Debridementandbone

graftingwithinternal ﬁxationviaanterior approach

Union

Zhangetal.[18] 2011 China Prospective 10 CervicothoracicTB withkyphosis

6:4 5.4±1.77 36(range,

26–47months)

One-stageposteriorfocus debridement,bonegraft fusion,and

instrumentation.

Spinaltuberculosiswas completelycuredinallten patients.Therewasno recurrenttuberculousinfection Ramanietal.[23] 2005 India Retrospective 61 SpinalTBaffecting

C3toD2

Median:32(7–68) 38(24–84) months

Patientswithinvolvement oftheC3-C6vertebrae underwentexcisionofthe involvedvertebraeand intervertebraldiscs followedbyreconstruction withtitaniumimplantsby anteriorapproach.A transclavicularapproach wasusedforpatientswith involvementoftheC7-D2 vertebrae.

Theneckpainscorebasedona visualanalogscalechanged fromapreoperativeaverageof 7to2atfollow-upafter4 months.52(85%)patienthad completereliefofpainwhile 16patientswhohadgradeIII toIVmusclestrengthregained completepower

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CASE REPORT – OPEN ACCESS

I.Salehetal./InternationalJournalofSurgeryCaseReports69(2020)109–113 113

Inadditiontoposteriorprocedures,strut graftormeshcage couldbeinsertedanteriorlytoprovideadditionalstability,espe- cially in our patient whose spinal biomechanics was heavily disruptedfromdestructionofmultiplevertebralsegmentsante- riorly.However,astheriskofgraftorcagefailureincreasedalong withtheincreasingnumberofinvolvedsegments,wedecidednot toperformanyprocedureanteriorly.Toprovideadditionalstability, weputthepatientonexternalbraceforsixmonths,afterwardsthe bracewasremovedgraduallyuntiltheinvolvedsegmentsregained bonyfusion.

5. Conclusions

Our report demonstrates one of the longest involvement of extensive contiguous CTSTB who was treated with one-stage posterior-onlyapproach.However,asthisisonlyareportofone case,furtherstudiesarerequiredtoinvestigatethesafetyandefﬁ- cacyofsuchapproachfortreatingextensiveCTSTB.

Sourcesoffunding Nonedeclared.

Ethicalapproval

EthicalapprovalhasbeenreceivedfromFatmawatiHospital, Jakarta,Indonesia.

Consent

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages.Acopy ofthewrittenconsentisavailableforreviewbytheEditor-in-Chief ofthisjournalonrequest.

Authorcontribution

IfranSaleh:performingtheprocedure,studyconcept,providing revisionstoscientiﬁccontentofthemanuscript.

DidikLibrianto:performingtheprocedure,datacollection,pro- vidingstylistic/grammaticalrevisionstothemanuscript.

PhedyPhedy:performingtheprocedure,dataanalysis,writing thepaper.

TotoSuryoEfar:datacollection,writingthepaper.

AnissaFebyCanintika:writingthepaper.

Registrationofresearchstudies

Thisstudyhasbeenregisteredat researchregistry.com(UIN:

researchregistry5169).

Guarantor IfranSaleh.

Provenanceandpeerreview

Notcommissioned,externallypeer-reviewed.

DeclarationofCompetingInterest None.

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