J. Coloproctol. (Rio J.) vol.36 número4

jcoloproctol(rioj).2016;36(4):244–247

w w w . j c o l . o r g . b r

Journal

of

Coloproctology

Case

Report

Low

digestive

hemorrhage

caused

by

a

vascular

malformation

in

a

patient

with

Crohn’s

disease:

case

report

and

literature

review

Carlos

Henrique

Marques

dos

Santos

_,

_José

_Valério

_Librelotto

_Stefanello

_II,

Yuri

Maciel

Carvalho,

Flávio

Renato

de

Almeida

Senefonte

UniversidadeFederaldeMatoGrossodoSul,CampoGrande,MS,Brazil

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t

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e

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n

f

o

Articlehistory:

Received10March2016 Accepted25April2016 Availableonline18June2016

Keywords:

Crohn’sdisease Complications Vascularmalformation

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Theauthorsreportthecaseofa19-year-oldmalepatientwithCrohn’sdiseasetreatedwith infliximabandazathioprine,withsustainedclinicalremission;inthecourseoftheevolution, thispatientshowedavascularmalformationinhisrectum,withdailymassivebleeding,in needofseveralbloodtransfusionsandfinallywithanindicationforsurgicaltreatment.Data fromtheliteratureontheassociationoftheinjuryatissueandCD,aswellasthedrugsused bythepatient,areevaluated.Theliteraturelacksinformationlinkingthismalformationwith CDorwiththedrugsused.

©2016SociedadeBrasileiradeColoproctologia.PublishedbyElsevierEditoraLtda.This isanopenaccessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/ licenses/by-nc-nd/4.0/).

Hemorragia

digestiva

baixa

por

malformac¸ão

vascular

em

paciente

com

doenc¸a

de

Crohn:

relato

de

caso

e

revisão

da

literatura

Palavras-chave:

Doenc¸adeCrohn Complicac¸ões Mal-formac¸ãovascular

r

e

s

u

m

o

Osautoresrelatamocasodeumpacientemasculinode19anosqueapresentadoenc¸a deCrohnemtratamentocomInfliximabeeAzatioprinacomremissãoclínicasustentada,o qualapresentaaolongodaevoluc¸ãoumamal-formac¸ãovascularnoreto,comsangramento diáriovolumoso,necessitandodediversastransfusõessanguíneasefinalmenteaindicac¸ão detratamentocirúrgico.Sãoavaliadososdadosdaliteraturaquantoàassociac¸ãodalesão apresentadaeaDCbemcomocomosmedicamentosemusopelopaciente.Nãohána literaturapesquisadanenhumainformac¸ãoqueassocieamal-formac¸ãocomaDCnem tampoucoosmedicamentosutilizados.

©2016SociedadeBrasileiradeColoproctologia.PublicadoporElsevierEditoraLtda.Este ´eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND(http://creativecommons.org/ licenses/by-nc-nd/4.0/).

∗ _{Correspondingauthor.}

E-mail:chenriquems@yahoo.com.br(C.H.Santos).

http://dx.doi.org/10.1016/j.jcol.2016.04.011

jcoloproctol(rioj).2016;36(4):244–247

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Introduction

Crohn’sdisease(CD)hasbecomeincreasinglycommoninour environment,andinaveryspecialwaythisconditionaffects youngindividuals. Dependingonthe mannerinwhich DC manifests itself, the disease can cause a departureof the patientfromhis/herdailyactivities,whichrepresentsgreat losses,bothpersonallyandsocially.

Amongthe various complications that the disease may cause,bowelbleedingisnotthemostfrequentand,whenit occurs,thiscomplicationtendstobebenign,providedthatan adjustmentofthetreatmentoftheinflammatoryprocessis carriedout.

Inthiscasereport,wedescribeanunusualsituation,that ofayoungmanwithDCinremissionwhocametouswith dailyrectalbleeding,aneventatfirstunderstoodasapossible recurrenceofthedisease.Buttheinvestigationfoundthatthe causeofthebleedingwasaconditionofvascularorigininthe rectum.Theaimofthisstudywastodeterminewhetherthere wassomerelationshipofthisvascularlesionwithDCorwith thedrugsinuse,orwhetheritwasjustacoincidence.

Case

report

A male patient, 19 years old, with a colonoscopy diagno-sis of CD established seven years ago, showing chronic, nonspecificileitisandcolitis;atthattime,aschemewith pred-nisone+infliximab(growthdeficit)wasinstituted.Thepatient showedclinicalimprovement;thus,prednisonewas discon-tinuedand,aftersixmonths,anewcolonoscopyshowedthat thediseasewasinremission.Thepatientcontinuedtheuse ofinfliximab.Fouryearsago,thepatienthadarelapse,when azathioprinewasprescribed,andanadjustmentofinfliximab dosewascarriedout,againwithagoodresponseand com-plete remission. About a year ago, the patient cameto us withanewlow bleedingpicture;the colonoscopyrevealed a rectum with an extensive purple/blue subepithelial area

Fig.1–Distalportionoftherectumwithoutvascularlesion.

Fig.2–Intermediateportionoftherectumwithvascular lesion.

suggestiveofischemiaorhemorrhage(Figs.1–3)withno evi-denceofanactiveinflammatorydisease.Then,thepatient wassubmittedtoaproctologyexaminationundersedation, and dilatedhemorrhoidal vesselswere foundin theentire analcircumference,withadistalrectumwithoutchange.A CTscanrevealedanabdomenwithmildthickeningand hypo-tonia ofiliacloops+thickening andhypotonia ofthedistal sigmoidcolonandrectum.Abowelarteriogramdemonstrated anarteriovenousmalformationintheupperandlower mesen-teric arteries (Figs. 4–6) without signs of ischemia. Taking intoaccountthatthebleedingpersistedforseveralmonths, requiringeightbloodtransfusions,itwasdecidedtoperform asurgery.Thepatientunderwentaproctectomywithcoloanal anastomosisandaprotectiveileostomy,withgoodevolution. Thehistopathologicexaminationconfirmedthatthecasewas

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Fig.4–MFVinuppermesentericartery.

oneofavascularmalformationwithoutdiseaseactivityinthe resectedmaterial.

Discussion

The occurrenceof lower gastrointestinal bleeding (LGB) in patientswithDCisaninfrequentcomplication.Lietal.1

evalu-ated1374patientswithactivedisease;5.31%showedLGB.The authorsnotedthattherecentuseofazathioprineandactive diseaseintheleftcolonwerethemainfactorsrelatedtothis outcome.Thepatientherereportedhadnoactivediseaseand wasusingazathioprineformorethanthreeyears.

ItiswellknownthatthemajorityofCDpatientsrequire surgicaltreatmentthroughouttheirlife,butbowelresections arealmostalwayspracticedforthetreatmentofactive dis-ease.Pengetal.2_{evaluatedthemainindicationsforsurgery}

Fig.5–MFVinuppermesentericartery.

Fig.6–MFVinintermediateportionoftherectum.

inpatientswithCD andfoundthatobstruction orstenosis rankedfirst(23.84%),followedbytherapeuticfailure(14.80%). Intestinalbleedingwasonlytheeighthmostfrequent indica-tion,with4.79%ofcases;atthispoint,itshouldbenotedthat notwithstandingthis finding,this complicationwasrelated todiseaseactivity,nottoassociateddisorders,e.g.,vascular malformation(asthatobservedinthepresentreport).

Someauthorshavereportedcasesofvascular malforma-tion in the rectum with frequentbleeding (as in the case here presented), and most of these patients also require surgicalresection.However,themedicalliteraturesurveyed lacksstudiesinwhichthisfact hasoccurred inindividuals withCD.3

Jabło ´nskaetal.4_{reportedacaseofvascularmalformation}

that occurredinapatientwithCD,butit wasacaseofan injuryonthepathofanenterocutaneousfistula.Theauthors emphasizedthescarcityofstudieswithsimilarcases,which leadsustobelievethatprobablythereisnorelationshipof DC withavascularmalformation.Thus,thequestion tobe answered iswhetherthetreatmentusedinthiscasecould beinsomewayassociatedwithvascularmalformation,since thiscomplicationaroseaftertheuseofthistherapy.

Sokumbietal.5_{reviewedcasesofvasculitisinCDpatients}

onanti-TNF andobservedaprevalenceofskinlesions,but theseauthorsalsoobservedsystemicandrenallesions,some ofthemcoursingwithbleedingepisodes.Severalother com-plications have been reported with the use of anti-tumor necrosisfactor(TNF)alphaantibodies,includinginterstitial lungdisease,skinlesions,opportunisticinfections,etc.; how-ever,nobowelvascularlesionwasidentified.6

jcoloproctol(rioj).2016;36(4):244–247

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Thus,it canbeconcludedthatneitherDC norits treat-ment appearstoberelated tothe onset ofbowel vascular malformation; however, considering the lack of articles addressing this issue, our report plays a big role, thanks to the documentation of the observed complication, as an eventual contribution to other publications that may arise.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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3.ChenZC,DengHJ.Laparoscopicdiagnosisandtreatmentof rectumtelangiectasia:reportofarareformoflower

gastrointestinalhemorrhage.NanFangYiKeDaXueXueBao. 2015;35:1508–10.

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Hemorrhagefromtheinferiorepigastricarterymalformation intotheexternalintestinalfistulaasanatypicalcomplication ofCrohn’sdisease.JCC.2013:e26–7.

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