Clinical and epidemiological aspects of American cutaneous leishmaniasis with genital involvement,

AnBrasDermatol.2020;95(5):641---644

Anais

Brasileiros

de

Dermatologia

TROPICAL/INFECTOPARASITARY

DERMATOLOGY

Clinical

and

epidemiological

aspects

of

American

cutaneous

leishmaniasis

with

genital

involvement

夽,夽夽

Marcelo

Rosandiski

Lyra

_,

_Alan

_Bittencourt

_da

_Silva

_,

Cláudia

Maria

Valete-Rosalino

_,

_Maria

_Inês

_Fernandes

_Pimentel

a_{LaboratoryforClinicalResearchandSurveillanceinLeishmaniasis,InstitutoNacionaldeInfectologiaEvandroChagas,Fundac¸ão}

OswaldoCruz,RiodeJaneiro,RJ,Brazil

b_{MedicalSchool,UniversidadeFederalFluminense,Niterói,RJ,Brazil}

Received25October2019;accepted8December2019 Availableonline15July2020

KEYWORDS Genitaldiseases, male; Leishmania braziliensis; Leishmaniasis, cutaneous; Leishmaniasis, mucocutaneous

Abstract Genitallesionsareanunusual presentationofAmericancutaneousleishmaniasis. ConditionssuchasdisseminatedcutaneousleishmaniasisandHIVinfectionmaybeassociated withgenitalinvolvement.TheauthorspresentfivecasesofAmericancutaneousleishmaniasis withgenitallesionsanddiscusstheclinicalandepidemiologicalaspectsobservedinthiscase series.

©2020SociedadeBrasileira deDermatologia.PublishedbyElsevierEspa˜na,S.L.U.Thisisan openaccessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

Introduction

American cutaneous leishmaniasis (ACL) is an infectious diseasecausedbyprotozoaofthegenusLeishmania trans-mitted by the bite of infected female sandflies, insects

夽 _{Howtocitethisarticle:LyraMR,SilvaAB,Valete-RosalinoCM,}

PimentelMIF.ClinicalandepidemiologicalaspectsofAmerican cuta-neousleishmaniasis withgenital involvement.AnBrasDermatol. 2020;95:641---4.

夽夽_{StudyconductedattheLaboratoryforClinicalResearchand}

Surveillance in Leishmaniasis, Instituto Nacional de Infectologia EvandroChagas,RiodeJaneiro,RJ,Brazil.

∗_{Correspondingauthor.}

E-mail:marcelolyradermato@hotmail.com(M.R.Lyra).

of the genus Lutzomyia.1,2 _{Clinically, ACL is divided into}

localizedcutaneousleishmaniasis,disseminatedcutaneous leishmaniasis (DL), diffuse cutaneous leishmaniasis, and mucosalleishmaniasis.1---3

DLconstitutesupto2%ofACLcasesandprobablyoccurs duetothelymphaticorhematicspreadoftheparasitefrom thebitesite.1_{Thisclinicalformischaracterizedbythe}

pres-enceofnumerous skinlesions,tenormore,distributedin twoor more non-contiguous body segments.3 _{Skin lesions}

arepolymorphicandtypicallyconsistofacneiformpapules, infiltratedor ulcerated plaques, warty lesions and ulcers withagranularbottomandraised edges.1,3_Verrucousand

vegetating lesions are rare.3 _{Systemic symptoms such as}

fever, myalgia, asthenia, and weightloss occur in 50% to 75% of cases;mucosal involvement, predominantly in the nasalmucosa,isobservedinupto53%ofDLcases.3,4

https://doi.org/10.1016/j.abd.2019.12.010

0365-0596/©2020SociedadeBrasileiradeDermatologia.PublishedbyElsevierEspa˜na,S.L.U.ThisisanopenaccessarticleundertheCC BYlicense(http://creativecommons.org/licenses/by/4.0/).

642 LyraMRetal. Genitallesionsarean unusualpresentation ofACLand

suggesthematicdisseminationinpatientswithDLordirect inoculationoftheparasiteinpatientswithisolatedgenital lesionswhosleepnaked outdoorsor performbodily func-tionsinendemicareasofACLwithoutsanitaryfacilities.5---7

OfHIVpatientswithACL,60%presentedDLand27%,genital lesions8

Case

reports

Table 1 describes the five patients with ACL with genital lesionstreatedbetween2007and2019,whocomprisedthe entire seriesof ACLwithgenital involvementobserved in this institution during this period. The mean age of the patientswas43years.AmongthosewithDL,alargenumber ofskin lesionswere observed,witha meanof 51 lesions. Themeantimefromtheonsetofgenitallesionsuntil diag-nosiswas5.6months.Thediagnosiswasconfirmedbyfinding theparasitesinoneor moreofthefollowingtests:direct examination(imprintorscraping),histopathological exam-ination, culture, and/or polymerase chain reaction (PCR) performedinbiopsiesoftheskin lesions.Allpatients pre-sented upper airway and digestive tract (UADT) mucosal

involvementand weretestedfor HIV andsyphilisinorder toruleoutco-infection.FourofthesepatientshadDLand twowereHIV-positive.Theglanswasthemostaffectedsite (Fig. 1). Four patients had painless penile ulcers, except for patient3,whohadpenileedema andmultiple painful lesionsontheforeskinthatpreventedtheexposureofthe glans.Aftertreatment,foreskinretractionallowed observa-tionofthelesionsontheglans(Fig.2).Patientfive(Fig.3) presentedanulcerationinthebodyofthepenisandanother inthescrotum.

Discussion

AlthoughSexuallyTransmittedInfections(STIs)arethemain causes of penile ulcers, other conditions such as fixed drugeruption erythema,autoimmune bullousdermatoses, psoriasis, Behc¸et’s disease, Reiter’s syndrome, pyoderma gangrenosum,lichen planus,andsquamouscellcarcinoma can also cause genital ulcers.9 _{ACL lesions are usually}

locatedin exposedareasofthebody,andgenital involve-mentisrarelyobserved.5---10_{PenilelesionsinACLareusually}

describedaspainlessulcerswithraisededgesandinsidious evolution, butextensive necroticulceration andkeratotic

Table1 ClinicalandepidemiologicalprofileofpatientswithACLwithgenitalinvolvement.

Patient 1 2 3 4 5 Ageinyears 41 47 65 43 27 HIV --- --- --- + + VDRL --- --- --- --- ---Numberofskin lesions 54 44 55 3 52 Presenceof mucosallesionsin UADT Nasalcavity, oropharynx Nasalcavity, nasopharynx Nasalcavity, oropharynxand larynx Oropharynx, nasopharynxand larynx Oralcavity Evolutiontime untildiagnosis

3months 4months 6months 6months 9months

Residence (City)

RiodeJaneiro Saquarema RiodeJaneiro RiodeJaneiro RiodeJaneiro Treatment Meglumine antimoniate Meglumine antimoniate Liposomal amphotericinB Liposomal amphotericinB Meglumine antimoniate

Americancutaneousleishmaniasiswithgenitalinvolvement 643

Figure2 (A),Patient3,withmultiplepainfululcersdistributedontheforeskinandbodyofthepenis.(B),Presenceof hyper-chromicscarsontheglansaftertreatment.

Figure3 Patient5,withpainlessulceratedlesionsinthe scro-tumandbodyofthepenis.

plaques have also been reported.5---8,10 _{Despite the small}

numberofpatientsinthisseries,itwasobservedthatthe following factors may be associated with genital involve-ment:mucosallesionsinUADT(100%);DL(80%),especially incaseswithalargenumberofskin lesions;andinfection byHIV(40%).Themostlikelyetiologicalagentinthisseries wasLeishmania(Viannia)braziliensiss,asallpatientswere inhabitants of the state of Rio de Janeiro withnorecent historyoftravel.1

ACL should be considered in the differential diagno-sis of chronic genital lesionsin patients whoreside in or travelfromendemicareas,especiallywhenassociatedwith mucosal lesions in UADT and multiple, polymorphic skin lesions.Furthermore,thepresenceofgenitallesionscanaid inthedifferentialdiagnosisofgranulomatousdiseaseswith similarclinicalpresentation,suchas paracoccidioidomyco-sis,histoplasmosis,anddisseminatedsporotrichosis.

Final

considerations

GenitalinvolvementinACLprobablyoccursduetohematic dissemination in patients with DL. Therefore, ACL should beincluded inthe differentialdiagnosis for patientsfrom endemicareaswithgenitalulcers,especiallyinthepresence ofmucosallesionsinUADTandmultipleskinlesions.

Financial

support

InstitutoNacionaldeInfectologiaEvandroChagas(INI).

Authors’

contributions

Marcelo Rosandiski Lyra: Approval of the final version of the manuscript; conception and planning of the study; elaborationandwritingofthemanuscript;obtaining, ana-lyzing,andinterpretingthedata;intellectualparticipation in propaedeutic and/or therapeutic conduct of studied cases;critical review of the literature; critical review of themanuscript.

Alan Bittencourt da Silva: Elaboration and writing of themanuscript;obtaining, analyzing,andinterpretingthe data;criticalreviewoftheliterature;criticalreviewofthe manuscript

Cláudia Maria Valete-Rosalino: Approval of the final version of the manuscript; elaboration and writing of themanuscript; intellectual participationin propaedeutic and/ortherapeuticconductofstudiedcases;criticalreview oftheliterature;criticalreviewofthemanuscript.

Maria Inês Fernandes Pimentel: Approval of the final version of the manuscript; elaboration and writing of themanuscript; intellectual participationin propaedeutic and/ortherapeuticconductofstudiedcases;criticalreview oftheliterature;criticalreviewofthemanuscript.

Conflicts

of

interest

Nonedeclared.

Acknowledgements

TheauthorswouldliketothankdoctorsAndréad’Ávila Fre-itas,Marcelo Luiz Carvalho Gonc¸alves, and Mayara Secco TorresdaSilva.

References

1.MinistériodaSaúde.SecretariadeVigilânciaemSaúde.In: Man-ualdevigilânciadaleishmaniosetegumentar.1rded.Brasília: MinistériodaSaúde;2017.

644 LyraMRetal. 2.Anversa L, Tiburcio MGS, Rochini-Pereira VB, Ramirez LE.

HumanleishmaniasisinBrazil:Ageneralreview.RevAssocMed Bras.2018;64:281---9.

3.MachadoGU,PratesFV,MachadoPRL.Disseminated leishma-niasis:clinical,pathogenic,and therapeuticaspects.AnBras Dermatol.2019;94:9---16.

4.Rosa MEA, Machado PRL. Disseminated leishmaniasis: clini-cal, immunological, and therapeutic aspects. DrugDev Res. 2011;72:437---41.

5.Cabello I,CaraballoA, MillánY. Leishmaniasisinthegenital area.RevInstMedTropSaoPaulo.2002;44:105---7.

6.Schubach A, Cuzzi-Maya T, Gonc¸alves-Costa CS, Pirmez C, Oliveira-NetoMP.Leishmaniasisofglanspenis.JEurAcad Der-matolVenereol.1998;10:226---8.

7.OsórioRC,BarbosaD,MartinsM,Leal R,NascimentoD, Fer-nandes E, et al. Tegumentary leishmaniasis (TL) caused by Leishmania Viannia braziliensis in genital organs. Gaz Med Bahia.2009;79Suppl3:91---4.

8.LindosoJA,BarbosaRN,Posada-VergaraMP,DuarteMI,Oyafuso LK,Amato VS, etal. Unusualmanifestationsoftegumentary leishmaniasisinAIDSpatientsfromtheNewWorld.BrJ Derma-tol.2009;160:311---8.

9.YesilovaY,TuranE,SürücüHA,KocarslanS,TanrikuluO,Eroglu N.Ulcerativepenileleishmaniasisinachild.IndianJDermatol VenereolLeprol.2014;80:247---9.

10.GülümM,Yes¸ilovas¸Y,Savas¸M,¸iftcC ¸i H,YeniE,etal.Acase ofgianthyperkeratoticcutaneousleishmaniasis inthepenis. TurkiyeParazitolDerg.2013;37:53---4.