Deep cutaneous ulcers and sinus formation in an immunocompetent adult,

AnBrasDermatol.2019;94(6):744---746

Anais

Brasileiros

de

Dermatologia

TROPICAL/INFECTOPARASITOLOGY

DERMATOLOGY

Deep

cutaneous

ulcers

and

sinus

formation

in

an

immunocompetent

adult

夽,夽夽

Qiang

Zhou

,

Kejian

Zhu

DepartmentofDermatology,SirRunRunShawHospital,ZhejiangUniversity,Zhejiang,China Received29July2018;accepted15December2018

Availableonline24October2019

KEYWORDS

Cryptococcosis; Infection; Skinulcer

Abstract Thisreportdescribes acaseofunusualdeepskinulcerswithtortuoussinustract formationinanimmunocompetentwoman.ShewasinitiallydiagnosedwithaStaphylococcus aureusskininfectionandhistopathologicallydiagnosedwithpyodermagangrenosum.However, culturefromthedeependofribbongauzeinsertedintothesubcutaneoussinustractrevealed shiny,light-yellowmucoidcolonies,whichwereidentifiedasCryptococcusneoformansvar. gru-bii.Shewastreatedwithfluconazoleforninemonthsandcompletelyhealed.Cryptococcosis isanopportunisticinfectioncausedbyvariantsofC.neoformansspecies.Cutaneous manifes-tationsofcryptococcosisarequitedivergent,rarelyoccurringasdeepskinulcerswithsinus formation.

©2019SociedadeBrasileiradeDermatologia.PublishedbyElsevierEspa˜na,S.L.U.Thisisan openaccessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

A43-year-oldwomandevelopedskinulcerswithslightpain andpurulentdischargeonherleftposterioraxillaryfoldfor fourmonthsandleftshoulderfortwomonths.Shehadbeen healthybefore, without underlying causes of immunosup-pression.She had hadnocontact withanimalsand birds. Physicalexaminationwasunremarkableexceptforthetwo deepcutaneousulcers(Fig.1).Serologicalexamswereall

夽 _{Howtocitethisarticle:ZhouQ,ZhuK.Deepcutaneousulcers}

andsinusformationinanimmunocompetentadult.AnBras Derma-tol.2019;94:744---6.

夽夽_{StudyconductedattheSirRunRunShawHospital,Schoolof}

Medicine,ZhejiangUniversity,Zhejiang,China.

∗_{Correspondingauthor.}

E-mail:3104093@zju.edu.cn(K.Zhu).

negative. Magnetic resonance imaging revealed soft tis-sueandmuscleinfectionwithsinusformation.Asinogram demonstratedtortuousirregularsinustractsextendingfrom thetwocutaneousulcers(Fig.2).Histologicalanalysis dis-played ulceration with dense infiltration of neutrophils. The repeated routinecultures found 100% Staphylococcus

aureus, while the patient was not responsive to antibi-otics.However,culturefromthedeependofribbongauze inserted into the sinus tract detected shiny, light-yellow mucoidcolonies(Fig.3).Indiainkstainshowed character-isticcapsulatedbuddingyeastcellswithhalos(Fig.4).The cerebrospinalfluidexaminationandbloodculturewere neg-ative. The biochemical and genetic identification proved that the isolatedpathogen wasCryptococcus neoformans

var.grubii.C.neoformansisusuallyrecoveredfromsoil con-taminatedwithavianexcreta,especiallypigeondroppings,

https://doi.org/10.1016/j.abd.2018.12.003

0365-0596/©2019SociedadeBrasileiradeDermatologia.PublishedbyElsevierEspa˜na,S.L.U.ThisisanopenaccessarticleundertheCC BYlicense(http://creativecommons.org/licenses/by/4.0/).

Deepcutaneousulcersandsinusformationinanimmunocompetentadult 745

Figure1 Ulcerontheleftshoulder.Thecutaneousulceron herleftshoulder,afteraskinbiopsy.

Figure2 Irregularsubcutaneoussinustracts.Asinogram illus-trating irregular sinus tracts extending from the cutaneous ulcers.

Figure 3 The cultured microorganism. Shiny, light-yellow mucoidcoloniesonSabouraud’sdextroseagar.

Figure4 Thespecialmicroorganismstain.TheIndiainkstain showedcharacteristiccapsulatedbuddingyeastcellswith dis-tincthalos(x40).

and decaying wood, fruits, vegetables, and dust.1

Cryp-tococcosisis anopportunistic fungal infectionthat occurs morecommonlyamongimmunocompromisedpatientswith acquired immune deficiency syndrome or other underly-ing diseases (e.g., diabetes mellitus, liver cirrhosis, and malignancies), and in subjects under immunosuppressive therapy.2,3 _{However, there are also reports of}

cryptococ-cosisin immunocompetent patients.4,5 _{Theauthors herein}

describeacaseofC.neofromansvar.grubiiinfection pre-senting as unusual deep skin ulcers with sinus formation but free of any clinical evidence of systemic diseases. In humans, C. neoformans causes three types of infec-tions: pulmonary cryptococcosis, cryptococcal meningitis, andcutaneouscryptococcosis.2_{Thoughconsideredasa}

dis-tinct clinical entity,1 _{cutaneous cryptococcosis is mostly}

believed to be attributed to inhalation of Cryptococcus spores and later hematogenous dissemination.2 _The

diag-nosisofcutaneouscryptococcosisisoftendifficultbecause the skin lesions are non-specific and have various clini-calmanifestations,suchascellulitis,plaques,ulcerations, pustules,granulomata,abscesses,andherpetiformor mol-luscumcontagiosum-likelesions.6_{However,almostanytype}

ofskinlesion---includingsuperficialskinulcers---canbeseen indisseminatedcryptococcosis,deepulcerswithsinustract formationareveryrareandhavenotyetbeenreported.It wastheradiographicimagesillustratingsofttissueinfection andexistenceofsinustractsthattriggeredtheauthors’ crit-icalideaofperformingaculturefromtheterminalportion oftheribbongauzeinsertedintothedeepsinustract,which finally produced a positive result for C. neoformans var.

grubii.The treatment for cryptococcosis depends on the anatomical site involved and immune status of the host. AccordingtotheguidelinesoftheInfectiousDiseases Soci-etyofAmerica,7 _{thepatientwastreatedwithfluconazole}

400mg daily for nine months, and a complete cure was observed.Thiscasehighlightsthatunusualdeepcutaneous ulcerswithsinustractformationmaysometimesbetheonly manifestationofdisseminatedcryptococcosisandshouldbe includedinthedifferentialdiagnosisofcutaneousulcerative lesions.

746 ZhouQ,ZhuK

Financial

support

This work was supported by the National Natural Science FoundationofChina(GrantNo.81573057).

Author’s

contributions

QiangZhou:Approvalofthefinalversionofthemanuscript; elaboration and writing of the manuscript; obtaining, analyzing and interpreting the data; effective participa-tion in research orientation; intellectual participation in propaedeuticand/ortherapeuticconductofthecases stud-ied;criticalreviewoftheliterature.

Kejian Zhu: Approval of the final version of the manuscript;criticalreviewofthemanuscript.

Conflicts

of

interest

Nonedeclared.

References

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