J. Coloproctol. (Rio J.) vol.37 número1

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jcoloproctol(rioj).2017;37(1):44–46

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

Journal

of

Coloproctology

Case

Report

Anorectal

hemangioma

–

differential

diagnosis

of

anal

bleeding

Walter

Batista

de

Santana

Neto

a

_,

_Giovanni

_Troiani

_Neto

a

_,

Carlos

Magno

Queiroz

da

Cunha

a

_,

_André

_Cavalcante

_Brasil

a

_,

_José

_Ney

_Primo

_Feitosa

b,c,∗

a_Universidade_de_Fortaleza,_Fortaleza,_CE,_Brazil

b_Sociedade_Brasileira_de_{Coloproctologia,}_Rio_de_Janeiro,_RJ,_Brazil c_Hospital_São_Raimundo,_Fortaleza,_CE,_Brazil

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Articlehistory:

Received28April2016 Accepted31August2016

Availableonline17September2016

Keywords:

Hemangioma Anorectal Diagnosis Surgery

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b

s

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Anorectalhemangiomaisoneoftherarestcausesoflowergastrointestinalbleeding,but isoftenneglectedandconfusedinthedifferentialdiagnosis.Theclinicalexaminationisa turningpointforacorrectdiagnosisandmanagementofpatients,thusavoiding unneces-saryprocedures.Thetreatmentofchoiceforthisconditionissurgicalandintraoperative bleedingisthemaincomplicationofthistherapy.Thepresentcasereportsa25-yearold patient witha historyof bleedingfromthe ageof13, beingdiagnosed withanorectal hemangioma,andsurgicallytreatedwithresectionoftheaffectedsegmentandwithwound synthesisbymarsupialization,withagoodprogressionpostoperatively.

Hemangioma

anorretal

-

diagnóstico

diferencial

de

sangramento

anal

Palavras-chave:

Hemangioma Anorretal Diagnóstico Cirurgia

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OHemangiomaAnorretal éumadasmaisrarascausasdeHemorragiadigestivabaixa, sendomuitasvezesnegligenciadaeconfundidanodiagnósticodiferencial.Oexameclínico representaumponto decisivoparacorretodiagnóstico emanejodopaciente,evitando realizaçãodeexamesdesnecessários,eotratamentodeescolhadessapatologiaécirúrgico, sendoosangramentointraoperatorioaprincipalcomplicaçãodessaterapêutica.Orelato decasoaseguirreportaahistóriadepacientecom25anos,queapresentavasangramento desdeos13,sendodiagnosticadocomHemangiomaAnorretaletratadocirurgicamentecom ressecçãodosegmentoafetadoesíntesedeferidacommarsupialização,evoluindobemno pós-operatório.

∗ _{Corresponding}_author_.

E-mail:joseneypf@hotmail.com(J.N.Feitosa).

http://dx.doi.org/10.1016/j.jcol.2016.08.001

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45 Introduction

AbleedingepisodewithitsorigindistaltotheTreitzangleis calledalowergastrointestinalbleeding;intheseconditions, theprevalenceachieves15%amongallgastrointestinaltract bleedings.Amongthecausesoflowergastrointestinal bleed-ing,about 95%emerge fromthelargeintestine.Inpatients under50years,thesebleedingsaremainlycausedbyinjuries ofanorectalregion,whileinthoseover70years,diverticular diseaseandangiodysplasiaarethemaincauses.1,2

Amongalloftheabovecauses,anorectalhemangiomais oneoftherarest,andoftenthisinjuryisneglectedinthe dif-ferentialdiagnosisandconfusedwithmostprevalentdiseases suchashemorrhoids, Crohn’sdisease,or ulcerativecolitis, causingdelays inthetreatment andmaybe causingsevere bleedingepisodesleadingtomortalityratesabove40%.3,4

Definedasabenigncongenitalvascularneoplasm, anorec-talhemangiomawasfirstdescribedin1839.5_Painless_rectal

bleedingisthemainclinicalmanifestation,andisoften asso-ciatedwithanemia.3,6–8

Diagnosticsuspicionarisesfromclinicalexaminationwith directvisualizationoftheinjury;thisexaminationcanbe com-plementedwithlaboratorytests,mesentericandinternaliliac arteriography,andcolonoscopy,dependingonthesite. Biop-siesshouldbeavoided,duetothehighriskofbleeding.3,9,10

Thetreatmentofchoiceissurgical,withresectionofthe affected segment, with preservation, wherever possible, of theanalsphincters.Amajorcomplicationistheoccurrence ofintraoperativebleeding,especiallyduringmoreextensive resections.3,4,8

Thefollowingcasereportdescribesthestoryofapatient withanorectal hemangioma who had bleeding complaints sincetheageof13,beingdiagnosedandtreatedsurgicallyby theageof25.

Case

report

Patient, 25 years, male, was admitted to a coloproctology service with the main complaint of‘bleeding through the anussincetheageof13′_(sic)._In_the_anamnesis,_the_patient

reportedthatsuchbleedingoccurredwithandwithoutregard to evacuation, increasing in intensity and becoming more frequent,sometimesincitinghemodynamicinstabilitywith hypotension,severepallor,andsyncope.Thissituationledthe patienttoseekanemergencyservice,inordertobe hospital-izedforbloodtransfusion.Thispatientreportedasthma in childhoodandreportedthathisfatherhadhemorrhoids.On physicalexamination,amucocutaneouspallor(++/4+), and apurplishstain inhisperianal region,aswell asaskinof spongy consistencyon digital rectal examination, between 4 and 7h, were found. The tests brought by the patient forthefirst consultationindicatedanemia (hemoglobin=7, hematocrit=21).Thepatientalsobroughtresultsofthe follow-ingstudies:high-digestiveendoscopy,colonoscopy,intestinal transit,endoscopiccapsule,andcomputedtomographyofthe abdomen;allthesetestswerewithinnormalityparameters.

Apelvic angioresonance was requested and performed, and wassuggestive of anorectalhemangioma. Thepatient

Fig.1–Exposureofananorectalhemangioma.

wasadmitted15daysaftertheconsultation,underwentblood transfusions tocorrecthisanemiaand wassubmittedtoa surgery.Duringtheprocedure,thehemangiomawasresected (Fig.1);theinjurywassituatedintheperianalregionandinto the canal.Thus, wedecidedinfavor ofamarsupialization ofthewound,inordertoreduceitssize,withaconsequent reductioninhealingtime.

Thepatient had agood progressionthrough the imme-diate postoperativeperiodand wasdischarged within48h. Histopathologicalexaminationofthesurgicalspecimen sup-portedafirmdiagnosisofhemangioma.Afollow-upscheme inthedoctor’sofficewasestablished,ateverytwoweeksfor thecasereview.Thewoundwascompletelyhealedinabout4 months.

Discussion

In the first place, inorder to obtain an effectivediagnosis inthecontextofahemangioma,itiscriticaltodifferentiate thetermsvascularectasia,angiodysplasia,andhemangioma, whichareoftenconfusedintheliterature.Thus,vascular ecta-siais aninjurywherethere occurs dilation ofpre-existing vessels;angiodysplasia isamalformation ofvesselsdueto adefectintheirformation,andhemangiomawouldbea neo-plasticinjury.11,12

Hemangiomas ofthe digestive systemoften are associ-atedwith skinlesionsofthe same nature.In thiscontext, despite the absence ofaknown etiology, insomecases, a familytendencyisfound,suggestinganautosomaldominant inheritance.3,4,11

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46

extensive, withan infiltrative or polypoidcharacter. When involvinga largersegment ofthe digestiveapparatus, cav-ernous hemangiomas characterize the so-called multiple phlebectasia.Bleedingistheirmostfrequentmanifestation, and this problem may begin in childhood with recurrent, of increasing severity, episodes; in addition, anemia is a constantfinding.(2)CapillaryHemangiomas,usuallysingle andasymptomaticformations,representing10%ofcolorectal hemangiomas;and(3)MixedHemangiomas,frequentlyfound inthestomach,smallintestine,andappendix.12

Thediagnosisofhemangiomamaybeconfusedwithmany otherdiseases,asalreadymentioned.Amesentericand inter-naliliac angioresonance, althoughnot conclusive in some cases,isaprocedureoftheutmostimportanceinthe diag-nosisofhemangioma.Evenwiththepossibilityofobtaininga diagnosis,onemustavoidusingbiopsies,duetothehighrisk ofbleeding.3,4,7,12

Thetreatmentofchoiceissurgical,througharesectionof theaffectedarea,buttherearesomeendoscopicand radio-logicalmethodsalreadypublishedthatcanbeeffective,such asethanolembolizationthrougharteriography.3,11,12

Thus, in view of the reported case, one must consider the critical importance that the anorectal hemangioma, althoughanuncommonneoplasm,shouldalwaystakepart inthedifferentialdiagnosisofanorectaldiseases,especially ina patientsufferingfrom lower gastrointestinal bleeding. Furthermore,itisworthemphasizingtheimportanceof con-ducting a thorough clinical examination that, per se, may suggest the diagnosis,guiding thephysicianin the correct propaedeuticstrategyforthesepatients.Thisprocedurewill avoidcostlytests,whichwillpostponethediagnosisandmay evenendangerthepatient’slife.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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