Rev. bras. ortop. vol.51 número6

rev bras ortop.2016;51(6):716–719

SOCIEDADE BRASILEIRA DE ORTOPEDIA E TRAUMATOLOGIA

w w w . r b o . o r g . b r

Case

Report

Intraosseous

anomalous

drainage:

a

rare

case

of

pretibial

varicose

vein

夽

Frederico

Barra

de

Moraes

_,

_Carolina

_Parreira

_Ribeiro

_Camelo,

_Marcelo

_Luiz

_Brandão,

Pedro

Ivo

Fávaro,

Tercília

Almeida

Barbosa,

Raul

Carlos

Barbosa

UniversidadeFederaldeGoiás(UFG),FaculdadedeMedicina,Goiânia,GO,Brazil

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r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received12October2015

Accepted13October2015

Availableonline28September2016

Keywords:

Varicoseveins

Drainage

Intraosseousinfusions/methods

a

b

s

t

r

a

c

t

Valvefailurewithrefluxandpost-thromboticsyndromearethefactorsmostcommonly

correlatedwithvaricosedisease.Otherrareetiologiescanbeputforwardwhenthesetwo

maincausesareruledout.Wereportacaseinwhichayoungmanpresentedchronicpainin

thelefttibia,varicoseveinsinthelowerlimbsandfrequentoccurrencesoferysipelas.

Dur-inginvestigationoftheetiologyofthevaricoseveins,radiographsandmagneticresonance

imagingoftheleftlegwererequested.Theseshowedimagessuggestiveofanosteolytic

lesioninthetibia,butledustothediagnosisofanintraosseousveinwithanomalous

drainage.ThiswasconfirmedthroughvascularexaminationscomprisingDopplervenous

flowmeasurementand phlebography.Recognition ofthisrareintraosseous anomalyis

fundamentalforproper patient management,but anintraosseoussurgicalapproach is

unnecessary.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublishedbyElsevierEditora

Ltda.ThisisanopenaccessarticleundertheCCBY-NC-NDlicense(http://

creativecommons.org/licenses/by-nc-nd/4.0/).

Drenagem

anômala

intraóssea:

um

caso

raro

de

varizes

pré-tibiais

Palavras-chave:

Varizes Drenagem

Infusõesintraósseas/métodos

r

e

s

u

m

o

Incompetênciavalvarcomrefluxoesíndromepós-trombóticasãoosfatoresmais

comu-menterelacionadosàdoenc¸avaricosa.Outrasrarasetiologiaspodemserlevantadasquando

essasduas principaiscausas sãodescartadas.Relatamos ocasodeum homemjovem

comdorcrônicanatíbiaesquerda,varizesemmembrosinferioreseerisipeladerepetic¸ão.

Duranteinvestigac¸ãodaetiologiadasvarizesforamsolicitadasradiografiaseressonância

dapernaesquerda,queevidenciaramimagenssugestivasdeumalesãoosteolíticanatíbia,

masquenoslevaramaodiagnósticodeveiaintraósseacomdrenagemanômala,confirmado

pelosexamesvascularesdedopplerfluxometriavenosaeflebografia.Oreconhecimento

夽

StudyconductedattheUniversidadeFederaldeGoiás,FaculdadedeMedicina,HospitaldasClínicas,DepartamentodeOrtopediae

Traumatologia,Goiânia,GO,Brazil.

∗ _{Correspondingauthor.}

E-mail:[email protected](F.B.Moraes).

http://dx.doi.org/10.1016/j.rboe.2015.10.014

2255-4971/©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublishedbyElsevierEditoraLtda.Thisisanopenaccessarticle

rev bras ortop.2016;51(6):716–719

717

dessararaanomaliaintraósseaéfundamentalparaomanejoadequadodopaciente,não

necessitadeabordagemcirúrgicaintraóssea.

©2016SociedadeBrasileiradeOrtopediaeTraumatologia.PublicadoporElsevier

EditoraLtda.Este ´eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND(http://

creativecommons.org/licenses/by-nc-nd/4.0/).

Introduction

Varicoseveins inthelower limbsare present in10–40%of

individualsaged30–70years.Theyareassociatedwith

condi-tionsthatimpairvenousreturn,suchaspregnancy,physical

inactivity,andclinicalpicturesofvenousinsufficiency.Their

mainpathophysiologicalsubstrateistheincreasedpressure

inthe peripheral venoussystem. Congenitalabnormalities

ofthe venous endothelial tissue and smooth muscle cells

canresultinthedilationofthevenouswall,withsecondary

valvularinsufficiency.However,thereisevidencetosupport

acquired valvular insufficiency.1,2 _{There are several causes}

ofvaricoseveinsinthe lower limbs.Theseincludevenous

insufficiencyinthesaphenofemoralorsaphenopopliteal

junc-tion or in the usual perforating veins. Varicose veins are

classified,accordingtotheir causes,asusual andunusual.

Amongtheunusualcauses,thefollowinghavebeendescribed:

vulvoperinealvaricosity,intraosseousperforatingvein

incom-petence, round ligament varicosity, persistent sciatic vein

incompetence, Klippel–Trenaunaysyndrome, and

portosys-temiccollateralpathways.Physiciansshouldbefamiliarwith

the full range of primary causes of varicose veins in the

lowerlimbsandtheirradiologicalmanifestations;theyshould

alsorecognizethecomplementaryroleofvenographyintheir

assessment.3,4

The recognition of this rare anomaly in intraosseous

venous drainage iscritical toproper patient management.

Thisstudyaimedtoreportararecaseofintraosseousvenous

drainage anomaly with pre-tibial varices in a patient who

presenteddifficult-to-diagnosechronicpainandlower limb

edemathatcoursedwithadecreaseinthepatient’squalityof

life.

Case

report

Malepatient,23yearsold,obese,residentofagarbagedump

inGoiânia, GO, Brazil,was attended toat the Orthopedics

emergencyroomoftheHospitaldasClínicas.Hepresented

with chronic pain atthe lower limbs, more severe on the

left,alongwithpainonpalpationofthemiddle-thirdofthe

tibia,associatedwithrecurrenterysipelas,butwithnosigns

ofinflammationatthatmoment,withasymmetricalchronic

edema at the left lower limb (LLL), suggestive of chronic

venousinsufficiencyandtruncularvaricoseveinsintheLLL

(Fig. 1). Hehad been attended atvarious services, without

receivingadiagnosisorspecifictreatment,whichlimitedhis

qualityoflife.Radiographsandcomputed tomography(CT)

oftheLLLshowedaradiotransparentlesioninthe

middle-thirdofthetibialshaft,withsignsofadjacentbonesclerosis

suggestiveofanosteolyticlesion(Fig.1).

Magneticresonanceimaging(MRI)disclosedanincreased

intraosseousvascularizationatthemiddle-thirdofthetibia,

withanomalousdrainageandpre-tibialvarices(Fig.2).Venous

Doppler ultrasound showed varicose veins throughout the

affectedlimb.Thepatientwasreferredtothevascularsurgery

department;surgicaltreatmentwasindicatedforcorrection

ofvenousdrainage,withresectionofvaricoseveinsandpain

relief.Anintraosseoussurgicalapproachwasnotnecessary.

Discussion

Boutinetal.1_{reportedthefirstcaseofintraosseousvarices}

in1997; sincethen,13 caseshavebeen reported. Allcases

presentedthesamecharacteristics,withpatientsbetween23

and75yearsandunilateralalterationswithsignsofvenous

insufficiency.Thepresentpatientfallsintothisagegroup,but

wasattendedatseveralserviceswithoutadefinitivediagnosis.

Thisisduetothelowprevalenceofthisinjury,whichmakes

diagnosisdifficult.

Allreportedpatientswereadultsbetween23and75years,

andmosthadpaininthelowerleg.Onlyonepatientdescribed

had intraosseous venous drainage anomaly at the fibula.3

Allotherreportedpatientshadintraosseousvenousdrainage

anomalyatthetibia.1–7

Fromaradiologicalstandpoint,themostimportantaspect

to determine the primary cause ofvenous insufficiency is

the presenceofveins with valvularincompetence and the

generalcharacteristicsofthevaricoseveins.VenousDoppler

ultrasound has the ability to demonstrate venous dilation

and valvular incompetence. In varicose veins with

com-plexmorphologyand/orunusualmanifestations,MRI,CT,or

venographycanbeusedforfurtherevaluation.CTvenography,

aswellasconventionalvenography,canbeusedto

demon-stratethepresenceofanenlargedintraosseousvein,whichis

connectedtothevaricoseveins.3,4

However, MRI may be the preferred method to confirm

intraosseousvenousdrainageanomaly,asitdoesnotuse

ion-izing radiation,is less invasive, and provides superior soft

tissue contrast.Furthermore,MRI canalsobe usedto rule

out othervascularanomaliesthatcouldrepresent

differen-tial diagnoses, such as arteriovenous malformation (AVM),

venous malformations, and hemangiomas.2,3 _{Notably, it is}

possible to differentiate varicose veins from intraosseous

venous drainageanomalythroughclinical aspects,because

venousmalformation typicallyoccursinchildhoodorearly

adulthoodandincreasesproportionallywiththechild’sage,

withoutregression.2

Thecauseandclinicalsignificanceofvaricoseveinswith

intraosseousdrainageanomalyarenotyetfullyunderstood.

Onehypothesisisthatanauxiliarydrainage,viaintraosseous,

718

Fig.1–(A)clinicalaspectoftheleftlowerlimbshowinglargeandtortuousvaricoseveins;(B)radiographicappearance;(C)

sagittalCTcutshowingaradiotransparentandosteolyticimage,withsignsofadjacentsclerosis.

casesofdeepvenousthrombosis).5_Kweeetal.4_reportedthe

caseofapatientwithapictureofunilaterallowerlimbvarices

originatedfromavenouscommunicationbetweenthe

periph-eralvenoussystemmemberandanintraosseousvein,causing

chronic pain ofthe affected limbassociatedwith varicose

veinsandsignsofchronicvenousinsufficiency.

The diagnosis of this injury can be achieved through

thecombinationofclinicalassessmentandimagingexams.

Radiographymayshowsignsofspinalsclerosisandcortical

defectsthroughoutthecommunicatingvesselpathway.ACT

canevidencethiscommunication;however,MRIisthemost

sensitiveandpreferredmethod,allowingforamoreprecise

diagnosisofthecommunicationsiteanditsimpact.

Venogra-phyisanauxiliarymethodthatallowsforadetailedstudyof

thevenoussystem.Ultrasoundwouldbethestandardmethod

forvascularevaluation,butthewavesarereflectedbyhigh

bonedensity,restrictingitsusetoDopplervelocimetryofthe

limb,whichdisclosesthesignsofvenousinsufficiencythat

thisconditioncancause.3,4

Intraosseousvenousdrainageleadstoanincreasein

pres-sure in theperipheral venoussystem, which consequently

culminatesinvenous valveimpairmentand signsof

insuf-ficiency. As a result, patients present massive varices and

an increased risk for thromboembolic events. Therefore,

Fig.2–Magneticresonance:(A)frontalcutinT1showingprominentvessel,hypointenseonthetopographyofthe

middle-thirdofthetibia;(B)perforatingveinthatentersthetibia,T2-hyperintense,sagittalcut;(C)numerousvaricoseveins

rev bras ortop.2016;51(6):716–719

719

surgical treatment ofvaricose veins should be established

by the angiology team; an intraosseous approach is not

necessary.6,7

Conflict

of

interest

Theauthorsdeclarenoconflictsofinterest.

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1.BoutinRD,SartorisDJ,RoseSC,PlechaEJ,BundensWP, HaghighiP,etal.Intraosseousvenousdrainageanomalyin patientswithpretibialvarices:imagingfindings.Radiology. 1997;202(3):751–7.

2.FlorsL,Leiva-SalinasC,MagedIM,NortonPT,MatsumotoAH, AngleJF,etal.MRimagingofsoft-tissuevascular

malformations:diagnosis,classification,andtherapy follow-up.Radiographics.2011;31(5):1321–40.

3.Díaz-CandamioMJ,LeeVS,GolimbuCN,ScholesJV,Rofsky NM.Intrafibularvarix:MRdiagnosis.JComputAssistTomogr. 1999;23(2):328–30.

4.KweeRM,KavanaghEC,AdriaensenME.Intraosseousvenous drainageofpretibialvarices.SkeletalRadiol.2013;42(6):843–7.

5.MiraultT,LambertM,VinckierL,LamotteC,CousynM,Hatron PY.Anomalousintraosseousvenousdrainage:ararecauseof pretibialvaricoseveins.JMalVasc.2010;35(6):373–6.

6.PehWC,WongJW,TsoWK,ChienEP.Intraosseousvenous drainageanomalyofthetibiatreatedwithimaging-guided sclerotherapy.BrJRadiol.2000;73(865):80–2.