Case for diagnosis. Keloidal cord-like lesion on the leg,

AnBrasDermatol.2020;95(3):386---389

Anais

Brasileiros

de

Dermatologia

WHAT

IS

YOUR

DIAGNOSIS?

Case

for

diagnosis.

Keloidal

cord-like

lesion

on

the

leg

夽,夽夽

Flaviano

da

Silva

Oliveira

_,

_Nadya

_Picanc

_¸o

_Lopes

_,

_Carolina

_Talhari

_,

Antonio

Schettini

a_{TropicalDermatologyClinic,Fundac¸ãoAlfredodaMattadeDermatologiaeVenereologia,Manaus,AM,Brazil} b_{DepartmentofDermatopathology,Fundac¸ãoAlfredodaMattadeDermatologiaeVenereologia,Manaus,AM,Brazil}

Received1April2018;accepted4July2019 Availableonline19March2020

KEYWORDS

Histology; Lacazia; Lobomycosis

Abstract Wereporta74-year-oldmalepresentedtoanoutpatientdermatologyclinicin Man-aus,Amazonas,withaone-yearhistoryofpruritic,keloidallesionsonhisleftlowerextremity. Histopathologyshowedroundstructuresinreticulardermis.Grocottmethenaminesilverstain revealednumerousroundyeastswiththickdoublewalls,occurringsinglyorinchainsconnected bytubularprojections.Thediagnosiswaslobomycosis.Althoughthekeloidallesionspresented bythispatientaretypicaloflobomycosis,theirlineardistributionalongtheleftlowerlimbis unusual.

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

Case

report

In 2005, a 74-year-old male presented with a one-year history of pruritic, cutaneous lesions on his left lower extremity.Physicalexaminationrevealedanerythematous plaqueandcord-like nodularlesionsonthe leftthighand

夽 _{Howtocitethisarticle:OliveiraFS,LopesNP,TalhariC,Schettini} A.Casefordiagnosis.Keloidalcord-likelesionontheleg.AnBras Dermatol.2020;95:386---9.

夽夽_{StudyconductedattheFundac¸ãodeDermatologiaTropicale} VenereologiaAlfredodaMatta,Manaus,AM,Brazil.

∗_{Correspondingauthor.}

E-mail:[email protected](C.Talhari).

leg. Culture, histopathological and mycological examina-tions were performed at that time but the patient was subsequentlylosttofollow-upuntil2017,whenhereturned withcomplaintsofrecurrent,secondarybacterialinfections ontheleftlowerextremity.Onphysicalexam,ulcerswith perilesionalinduration,erythema,anddesquamationwere notedontheleftleg(Fig.1A).Additionally,cord-like,firm, brown,noduleswereobservedalongthemedialaspectofhis leftthighandleg(Fig.1B).Routinelabswereunremarkable.

Whatisyourdiagnosis?

a) Lymphangiticcutaneousleishmaniasis; b) Nontuberculousmycobacteriosis; c) Lobomycosis;

d) Sporothricosis.

https://doi.org/10.1016/j.abd.2019.07.008

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

Keloidalcord-likelesionontheleg 387

Figure1 Ulcerswithperilesionalhardeningoftheskin,erythemaanddesquamationwereseenontheleftleg(A);cord-like, hard,brownishnodularlesionswerealsoobservedalongthemedialaspectofhisleftthighandlegatphysicalexamination(B).

Figure2 (A)Biopsysite(arrow).(B)Histopathologydemonstratedhyperkeratosisandacanhosis,collagenfibroplasiavascular, neoformationanddiffuseinflammatoryinfiltrate consistingoflymphocytes,epithelioid cells,giantcellsandhemosiderin-laden histiocytesinthepapilardermis(Hematoxylin&eosin,×100).

Cutaneous biopsy was performed on the ulcer border (Fig. 2A). Histopathology showedy showed hyperkeratosis and epidermal acanthosis with a diffuse, predominantly lymphohistiocyticinfiltrate.Giantcells,hemosiderin-laden histiocytesandincreasedvascularityarenotedinthe pap-illary dermis (Fig. 2B). Round structures are present in reticulardermis (Fig.3A),andGrocott’smethenamine sil-verstainrevealsnumerousroundyeastswiththickdouble walls, occurring singly or in chains connected by tubular projections(Fig.3B).

Diagnosis:Lobomycosis.

Discussion

Lobomycosis, also known as Jorge Lobo’s disease and lacaziosis,isasubcutaneous,chronic,granulomatous myco-sis initially described by Jorge Lobo, in 1931. There are no systemic manifestations in lobomycosis.1,2 _{Due to the}

similarityofitsetiologicalagent,Lacazialoboi,to

Paracoc-cidioidesbraziliensis,themycosishasalsobeendescribed

askeloidalblastomycosis.3_{Lacazialoboihasneverbeen}

iso-latedin culturefromhumansor animals. Sincetheinitial description of lobomycosis in patients from the Brazilian

388 OliveiraFSetal.

Figure3 (A)Roundstructuresinreticulardermis(Hematoxylin&eosin,×200).(B)Grocott’smethenaminesilverstainrevealed numerousround yeastsarranged containingthick double walls, occurringsingly orin chainsconnected by tubular projections (Groccott,×400).

Amazon region, other cases have been reported in tropi-calandsubtropical regionsofLatin Americancountries.2,4

The few lobomycosis reports from outside these regions haveoccurredpredominantlyinpatientswhohadtraveled throughendemic areas. In 2008, cases werediagnosed in SouthAfricainpatientswithnohistoryoftravellingtoknown endemicareas.5

An important fact relatedto thismycosis was the dis-covery, in the Florida coast in 1970, of a dolphin with a clinical and histopathological disease similar to human lobomycosis.2,6_{Subsequently,otherdolphinswiththesame}

diseasewerefoundintheAtlanticcoastoftheUnitedStates, coastalregionof LatinAmericanandCaribbeancountries, coastalregionoftheSouthernBrazilianstatesofRioGrande doSulandSantaCatarina,PacificcoastandIndianOcean.7

There is only one documented case of dolphin to human transmission of lobomycosis: an aquarium attendant who worked in an aquariumpool with an infected bottlenose dolphin(Tursiopstruncatus)caughtinBayofBiscay,Spain.8

The disease is clinicallycharacterized bythe presence ofnodular, verrucous or keloidallesions,which arefound aloneorinplaques,localizedordisseminated,witha gener-allylongevolution.Themostcommonclinicalpresentation is keloidal,characterizedby firm, shiny, pinkish-brown to brownnodules.2 _{Ulceration mayoccurin areas subjectto}

trauma.Developmentofsquamouscellcarcinomain long-standinglesionsmayoccur.9

Anotherclinicalaspectthatdrewattentioninthispatient was the linear arrangement of lesions along the leg and thigh.Itisadmittedthatthespreadofthediseaseoccursby contiguity,andthroughlymphaticroute;however,anaspect similartothatobservedinthepresentcaseisnotcommon.

Financial

support

Nonedeclared.

Authors’

contributions

FlavianodaSilvaOliveira:Intellectualparticipationinthe propaedeutic and/or therapeutic conduct of the studied cases.

Nadya Picanc¸o Lopes: Obtaining, analysis, and inter-pretation of the data; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases.

Carolina Talhari: Approval of the final version of the manuscript; conception andplanning of the study; elabo-ration and writing of the manuscript; obtaining, analysis, and interpretation of the data; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; criticalreview of themanuscript.

Antonio Schettini: Approval of the final version of the manuscript;conceptionandplanningofthestudy; elabora-tionandwritingofthemanuscript;intellectualparticipation inthepropaedeuticand/ortherapeuticconductofthe stud-iedcases;criticalreviewoftheliterature;criticalreviewof themanuscript.

Conflicts

of

interest

Nonedeclared.

References

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Keloidalcord-likelesionontheleg 389

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