J. Coloproctol. (Rio J.) vol.35 número3

jcoloproctol(rioj).2015;35(3):175–177

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

Journal

of

Coloproctology

Case

Report

Endoanal

pilonidal

sinus:

case

report

and

literature

review

Carolina

Talini

_,

_Geanine

_Baggio

_Fracaro

,

Allan

Cezar

Faria

Araújo

,

Doryane

Maria

dos

Reis

Lima

,

André

Pereira

Westphalen

a_{HospitalUniversitáriodoOestedoParaná(HUOP),Cascavel,PR,Brazil} b_{SociedadeBrasileiradeColoproctologia,RiodeJaneiro,RJ,Brazil}

c_{UniversidadeEstadualdoOestedoParaná(UNIOESTE),Cascavel,PR,Brazil} d_{FaculdadeAssisGurgacz,Cascavel,PR,Brazil}

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Articlehistory:

Received2February2015 Accepted20February2015 Availableonline29April2015

Keywords:

Pilonidalsinus Rectalfistula

Transrectalultrasound

a

b

s

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PilonidalsinusisatermfirstusedbyHodgesin1880todescribegranulomatouslesions containinghairsinitsinterior.Thepresenceofendoanalpilonidalsinusisrareandonly ninecaseshavebeenreportedinmedicalliterature.Thisarticledescribesamale,42years, initiallysubmittedtofistulotomywithsetonplacementthatfouryearslaterevolvedwith complaintsofanalpurulentdischargeassociatedwithpainfulanalgroin.Duringthefirst evaluationasmallendoanaltumorwasfound.Itwaslocatedinposterolateralanusposition andcontainedhairinitsinterior,withoutpurulentdischargeatthatmoment.Underwent surgerythatconfirmedthepresenceofendoanalpilonidalsinus.Thesinuswasopened andlefttohealbysecondaryintention.Thepatienthadgoodoutcomewithnosignsof recurrence.

©2015SociedadeBrasileiradeColoproctologia.PublishedbyElsevierEditoraLtda.All rightsreserved.

Cisto

pilonidal

endoanal:

relato

de

caso

e

revisão

de

literatura

Palavras-chave:

Cistopilonidal Fístulaanal Ultrassomendoanal

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e

s

u

m

o

Otermo cistopilonidalfoi descritopela primeiravezpor Hodgesem1880 para descr-everlesõesgranulomatosascontendopêlosemseuinterior.Apresenc¸adecistopilonidal endoanaléraraeexistemapenasnovecasosrelatadosnaliteraturamédica.Descreve-se umcasomasculino,42anos,emacompanhamentohá5anosnoservic¸o.Submetido inicial-menteàfistulotomiaemdoistemposcomsedenho,evoluindo4anosdepoiscomqueixasde secrec¸ãopurulentaintermitenteporviaanalassociadaapresenc¸adetumorac¸ãodolorosa. Noexamefísicoconstatou-sepresenc¸adeorifíciocontendopêlosemseuinterior,sem

∗ _{Correspondingauthor.}

E-mail:andrewestphalen@terra.com.br(A.P.Westphalen).

http://dx.doi.org/10.1016/j.jcol.2015.04.002

176

secrec¸ãopurulentanomomentodoexame.Foiencaminhadoparatratamentocirúrgicoque confirmoudiagnósticodecistopilonidalendoanal.Noprocedimentofoirealizadaabertura docistoquefoideixadoparacicatrizarporsegundaintenc¸ão.Opacienteapresentouboa evoluc¸ão,semsinaisderecidivalocal.

©2015SociedadeBrasileiradeColoproctologia.PublicadoporElsevierEditoraLtda. Todososdireitosreservados.

Introduction

Pilonidal sinus is a term first used by Hodges in 1880 to describe granulomatous lesions containing hairs in its interior.1_{SuchlesionswerefirstdescribedbyWarrenin1854}

andcalledhairycyst.2_{Isacommonentityinyounghairyman}

andmostofthemcontainlonghairwithnofollicles.1

Similar lesions are being described inother body parts suchasabdominalwall,ears,hands,armpit,gluteal, interdig-italregion,nipple,occipital,perineumandumbilicus.1–3_This

diversityoftopographyisafactorthatsupportsatheory,and insupportofthattheory,someauthorssuggestthatpilonidal cystsaretheresultoflosthairsthatarepushedintotheskin. Thepresenceofendoanalpilonidalsinusisrare4_andonly

ninecaseshavebeenreportedinmedicalliterature.5_Thereare

severalwaystoexplainthegenesisofthosesinusintheanal canal.Oneofthemregardsanextensionofasacro-coccygeal cysttotheperianalregionandotherlesswidespreadtheories arethatthepilonidalcystcouldbebornthroughtheentryof hairsintoanopenanal fissureorthathaircangetintothe analcanalregionastheydoelsewhereinthebody.1,3,4

Nowa-daysthemostacceptedtheoryconcernstheacquireddisease afterperforminganalfistulotomyorothersurgicalprocedure inwhichhairmayenterthesubcutaneoustissuethroughthe healingwoundor throughthenewlyformedscar andmay serveasthecradleofapilonidalsinus.1,4,5

The pilonidal sinus can often be confused with anal fistula. Usually the patient has multiple recurrences and underwentseveralsurgicalproceduresbutwithoutsymptoms improvement.6

Thisarticleaimstoreportanendoanalpilonidalsinuscase thatappearedaftersurgicaltreatmenttocorrectananal fis-tula.

Clinical

presentation

GF,male,42years,smoker,diagnosedwithHIV10yearsago (usingEfavirenz,LamivudineandZidovudine),whofirstcame tothisColoproctologyservice5yearsago,complainingabout afastgrowinganaltumor,painful,whichevolvedto sponta-neousdrainageofpus.Afterfirstserviceadmissionananal posterolateralfistulawasdiagnosedinassociationwithgrade Ihemorrhoidsand multipleperianalskintags. Thepatient underwentsurgicalprocedurewithexcisionofskintags(that pathologicalanatomyconfirmedthatitwassquamous papil-loma with hyperkeratosis associated with viral cytopathic default),fistulotomywithsetonplacementandcauterization ofhemorrhoidalnipples.

Fig.1–3Dendoanalultrasoundshowingthinningofthe analcanal,withoutanylesions.

Aboutayearagostartedpresentinganorectalcomplaints again.Relateddrainageofpurulentandfetidsecretion dur-ingthisperiodandpainduringevacuationwithoutbleeding, denyingincreasingonthenodulesize.Duringthefirst eval-uationasmallendoanaltumorwasfound.Itwaslocatedin posterolateralanuspositionandcontainedhairinitsinterior, withoutpurulentdischargeatthatmoment.

Patient was submitted to 3D endoanal ultrasound that found no lesions despitethe thinning ofthe left dorsolat-eralinternalandexternalanalsphincterintheposterolateral quadrant ofthemiddle anal canal(Fig. 1). Itwasclinically diagnosedaspilonidalsinusoftheanalcanalandthepatient underwentsurgery.

jcoloproctol(rioj).2015;35(3):175–177

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Fig.2–Transoperatorydemonstratingposterolateralsinus containinghairinitsinterior.

Discussion

Differentpilonidalsinuspresentationsaredescribedin litera-ture.Regardingthoselocatedinendoanalregion,eightcases werereportedinmanandoneoftheminawoman.1–5_When

itcomestotheage,thosewereallyoungpatients–23,27,29, 30,42,46,55and58yearsforthemenand42yearsforthe woman.

Fourofthempresentedrecurrentpurulentdischargeand weresubmittedtothemostdiversesurgicalprocedures with-outclinicalimprovement.1,3–5_{Twoofthemhadnosymptoms}

and were diagnosedduringa surgicalprocedure to correct hemorrhoids4 _{andthreeofthemweresymptomaticforthe}

firsttime.2_{Noneoftheevaluatedpatientshadfoundhairin}

endoanalregionbythemselves.Asinthepresentedcase,the sinuswereuniqueineightofthereportedcasesandoneother patientpresentedtwoendoanalsinus.1

When it comesto the surgicalprocedure, infour cases thecysttrajectorywasopenedandlefttohealbysecondary intention1,4_{justlikethecasepresentedhereandontheother}

fourcasesthelesionwascompletelyremoved.2–5

Oneofthereportedcases5_{hadasimilaroutcomeastheone}

wepresenthere.Bothpatientswereinitiallysurgicallytreated tocorrectananalfistulawithseton,evolvedwithsymptoms recurrenceanddevelopedendoanalpilonidalsinus.

Oneofthepreviouscasereportshadatwoyearsfollow-up4

and none of the five patients presented disease recur-rence. Other author presentedoneyear follow up with no recurrence.5_{Allothercasereportspresentednoinformation}

aboutthefollow-upperiod.1–3

Conclusion

Endoanalpilonidalsinusisarareentitywithveryfewreported casesinliterature.Intherelatedcasesmostpatientsareyoung menwhoaresymptomaticforalongtimeuntilthetrue dis-easeisdiagnosed.Surgicaltreatmentseemstobeeffectivefor healingwithlowrecurrencerates.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

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1.WilsonE,FailesDG,KillingbackM.Pilonidalsinusesoftheanal canal:reportofcase.DisColonRectum.1971;14(6):468–70.

2.WestonSD,SchlachterIS.Pilonidalcystoftheanalcanal:case report.In:ReadatthemeetingoftheAmericanProctologic Society.1962.

3.AccarpioG,DaviniMD,FazioA,SenussiOH,YakubovichA. Pilonidalsinuswithananalcanalfistula:reportofcase.Dis ColonRectum.1988;31(12):965–7.

4.OrtizH,MartiJ,DeMiguelM,CarmonaA,Caba ˜nasIP. Hair-containinglesionswithintheanalcanal.IntJColorectal Dis.1987;2:153–4.

5.AlrawashdehW,AjazS,HammondTM,PorrettTRC,LunnissPJ. Primaryanalpilonidaldisease.ColorectalDis.2008;10:303.