J. Coloproctol. (Rio J.) vol.37 número3

jcoloproctol(rioj).2017;37(3):238–241

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

Journal

of

Coloproctology

Case

Report

Perianal

leiomyoma

夽

Bruna

Fernandes

dos

Santos

,

Lucas

Rodrigues

Boarini

,

Pietro

Dadalto

Oliveira

,

Fernanda

Belotti

Formiga

_,

_Galdino

_José

_Sitonio

_Formiga

_,

Anderson

da

Costa

Lino

Costa

a_{HospitalHeliópolis,Servic¸odeColoproctologia,SãoPaulo,SP,Brazil}

b_{HospitalHeliópolis,Servic¸odeAnatomiaPatológica,SãoPaulo,SP,Brazil}

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Articlehistory:

Received18September2016 Accepted27March2017 Availableonline10May2017

Keywords:

Gastrointestinalneoplasia Gastrointestinalstromaltumors Leiomyoma

Analcanal

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Leiomyomasaresmoothmuscletumorsandmayoccurinplaceswherethesefibersare present,whiletheanorectallocationisrare.Theyarecommonlyincidentalimagingfindings andinmostcases,patientsareasymptomatic.Thetherapeuticrecommendationistumor resectionandpostoperativefollow-up.Casereport:a38-year-oldBlackwomanhad,one yearago,aswellinginperianalrightregion,whichshowedslowandprogressivegrowth.She deniedbowelhabitalterations,localpain,hematochezia,ortenesmus.Proctologic exami-nationshowedafibroelastic,regular,mobile,painlessnodulemeasuring10cmatitslargest diameterintherightperianalregion,nexttotheanalverge.Thesofttissueultrasoundimage identifiedasolid,hypoechoic,anddiscreetlyvascularizednoduleintheperianal, superfi-cialrightglutealregionthatdidnotreachtheadjacentmuscles.Acompleteresectionof perinealtumorwascarriedoutintheventralposition.Histologicaland immunohistochem-icalanalysesdisclosedaleiomyomawithapositivefindingforactinsmoothmuscleand negativefordesmin.Sheiscurrentlyasymptomaticandundergoingoutpatientfollow-up.

©2017SociedadeBrasileiradeColoproctologia.PublishedbyElsevierEditoraLtda.This isanopenaccessarticleundertheCCBY-NC-NDlicense(http://creativecommons.org/ licenses/by-nc-nd/4.0/).

Leiomioma

de

localizac¸ão

perianal

Palavras-chave:

Neoplasiasgastrointestinais Tumoresdoestroma gastrointestinal Leiomioma Canalanal

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Osleiomiomassãotumoresdamusculaturalisapodendoocorrernoslocaisonde essas fibrasestãopresentes,sendoraraalocalizac¸ãoanorretal.Namaioriadoscasosospacientes sãoassintomáticos,sendocomumenteachadosdeexamedeimagem.Arecomendac¸ão terapêuticaéaressecc¸ãotumoraleoseguimentopós-operatório.Relatodocaso:mulher,38 anos,negra.Háumano,apresentouabaulamentoemregiãoperianaldireita,decrescimento lentoeprogressivo.Negavaalterac¸ãodohábitointestinal,dorlocal,hematoquezia,puxoou

夽

StudycarriedoutatHospitalHeliópolis,Servic¸odeColoproctologia,SãoPaulo,SP,Brazil.

∗ _{Correspondingauthor.}

E-mail:[email protected](B.F.Santos).

http://dx.doi.org/10.1016/j.jcol.2017.03.003

jcoloproctol(rioj).2017;37(3):238–241

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tenesmo.Aoexameproctológico,apresentavanodulac¸ãofibroelástica,regular,móvel, indo-lor,com10cmdediâmetroemregiãoperianalàdireita,próximaàbordaanal.Realizou ultrassonografiadepartesmolesqueidentificouimagemnodular,sólida,hipoecogênicae discretavascularizac¸ãoempartesuperficialperianaleglúteadireita,nãoenvolvendo mus-culaturaadjacente.Foisubmetidaàressecc¸ãocompletadotumorviaperineal,emposic¸ão ventral.Olaudohistológicoeimuno-histoquímicorevelouleiomioma,comachadopositivo paraactinademúsculolisoenegativoparadesmina.Atualmenteestáassintomática,em seguimentoambulatorial.

©2017SociedadeBrasileiradeColoproctologia.PublicadoporElsevierEditoraLtda.Este ´eumartigoOpenAccesssobumalicenc¸aCCBY-NC-ND(http://creativecommons.org/ licenses/by-nc-nd/4.0/).

Introduction

Leiomyomasaretumorsthatoriginatefromthesmooth mus-cleandmayoccuratthesiteswherethesefibersarepresent.1

Inthedigestive tract,themostcommon presentationisin thestomach,followedbythesmallintestine,beingunusually foundintheanorectalregion,wheretheyrepresentlessthan 0.1%ofthetumorsoftherectum,withararepresentationin softparts,mainlyintheperianaltopography.2–4

Intheliterature,theperianal leiomyomafinding israre: isolatedcaseshavebeendescribed,and themostcommon presentationisapainlesstumorinthislocation.2

To illustrate such condition, the authors report a well-documentedcaseanddiscussclinicalaspectstogetherwith literaturedata.

Case

report

A38-year-oldBlackfemalepresentedwithaswellinginthe rightperianalregion,ofslowandprogressivegrowthsincea yearbefore.Shedeniedchangesintheintestinalhabits,local pain,hematochezia,feelingofincompletedefecationor tenes-mus.Attheproctologicalexamination,shehadafibroelastic, regular,mobile,painlessnodulewith10cmindiameterinthe rightperianalregion,neartheanalverge.Shewassubmitted toasofttissueultrasonography,whichidentifiedanodular, solid,hypoechogenicanddiscretelyvascularizedimageinthe superficialperianaland rightglutealarea,notreachingthe adjacentmusculature.

In the ventral position, she underwent local anesthe-siawithsedation andcompleteresectionofperianaltumor (Figs. 1 and 2).The histologicaland immunohistochemical reportdisclosedaleiomyoma,withapositivefindingforactin andnegativefordesmin(Fig.3).

Discussion

Firstdescribed byVirchow in1854, and histologically con-firmedbyMalassez,in1872,leiomyomasarebenigntumorsof mesenchymaloriginandcandevelopwherethesmooth mus-cleispresent.3,5_{Theyaremorecommoninthefemalegenital}

tractandskin.Intheanorectalregion,theyrepresentonly3% ofallleiomyomasofthegastrointestinaltractandlessthan 0.1%ofrectumtumors,rarelyfoundinsofttissues,mainlyin

Fig.1–Patientintheventralpositionwithtumorextrusion fromtheperianalregionontheright,neartheanalverge.

theperianal area,wheretheincidenceis3.8%ofallbenign tumorsofsofttissues.6,7

Historically, gastrointestinal mesenchymal tumors were called benign (leiomyoma) or malignant (leiomyosarcoma). Recently, gastrointestinal stromal tumors(GIST) have been considered the best terminology. They are differentiated by immunohistochemical staining, in which a leiomyoma exhibits positivity for smooth muscle actin and negativity for CD117 and CD34 (c-Kit). Desmin may be absent or be expressedlater.5,8 _{Histologicalpatternsare similartothose}

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Fig.2–Surgicalspecimenaftertumorresection.

increasednumberofmitoses(>2mitosesperfield,witha10×

increase).3,9

Theliteraturehasscarcereportsofperianalleiomyomas: isolatedcasesaredescribed,andthereisnospecificsymptom, butwhenpresent,themostcommonpresentationisa pain-less perianal tumor. Other clinical manifestations include: localpainassociatedornotwithdefecation,foreignbody sen-sation,altered intestinal habits and rectal bleeding, which vary accordingtothe size,location and directionoftumor growth.3,10

Theyareclassifiedintotwomainvariants:superficial, rep-resented mainly by angioleiomyoma of the female genital tract;and deep, initially described by Kilpatrick et al. and Billingsetal.in1994and2001,respectively.Itissubdivided into somatic, which affects both genders and extremi-ties (mainly the thigh) and retroperitoneal, which occurs

preferentiallyinwomenduringthemenopauseperiod,ina pelvicretroperitoneallocation.4,5

Preoperative diagnosisis difficultbecause mostpatients are asymptomatic,beingcommonlyidentifiedduring endo-scopicandimagingexaminations.Biopsyisoftennot infor-mative,becauseitdoesnotinvolvetheentiretumor,impairing thefullevaluationofthehistologicalcharacteristics.8,11_Every

perianaltumorneedsadefinitivediagnosis,giventhe impor-tanceofadifferentialdiagnosiswithmalignanttumorsofthe rectum, anal and perianal canal,suchas: leiomyosarcoma, GISTs, liposarcomas, fibrosarcomas and carcinoids. There-fore,aproctologicalexaminationshouldbeperformed, and complementary examinations, such as perianal ultrasono-graphy ofsoft or endoanal tissues,computed tomography, magnetic resonance imaging and colonoscopy may be used.8,10,12

Thetreatmentoftheperianalleiomyomaconsistsinthe complete surgical resection, ensuring tumor-free margins, withtheexcisionalbiopsybeingdiagnosticandtherapeutic, since the histological analysis is necessary for its confir-mation.Anteriorresectionorabdominoperinealamputation shouldbeperformedincaseswherelocalresectionis impos-sible due totumor size or sphincter muscle infiltration.3,4

Therecurrencerateislowwhencomplete localexcisionis performed, but the extended postoperative follow-up with clinical and complementary examinations is important to confirmtheabsenceofthediseaseortodetectanyrecurrences and/ormalignanttransformation.3,5,10

Thepatientshowedswellingintherightperianalregion, painless,withnootherassociatedsymptoms,similartomost ofthepresentationsdescribed byother authors.2,3,8,10 _After

proctologicalexaminationandsofttissueperianal ultrasono-graphy,shewassubmittedtocompletetumorresectionand postoperativefollow-up,asrecommendedintheliterature.

Final

considerations

All perianal lesions need a definitive diagnosis. Excisional biopsy is the gold standard for the circumscribed tumor lesionsandthefindingofaleiomyomaisrareinthis topogra-phy.

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Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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8.CamposFG,LeiteAF,AraújoSEA,AtuíFC,SeidV,Habr-Gama A,etal.Anorectalleiomyomas:reportoftwocaseswith differentanatomicalpatternsandliteraturereview.RevHosp ClinFacMedSPaulo.2004;59:296–301.

9.SaundersRN,PattendenC,AgarawalPK.Heavyrectal bleedingsecondarytothepassageofarectalleiomyomaper anus.AnnRCollSurgEngl.2004:2004.

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