Chromoblastomycosis is a neglected tropical disease typically found in endemic tropical and subtropical regions. Herein, we discuss a rare case of a 55-year-old man in Texas who presented with an exophytic papule on the forearm, diagnosed to have chromoblastomycosis by shave biopsy and subsequent histopathological analysis. Treatment options for chromoblastomycosis include long-term oral antifungal therapy with itraconazole, physical modalities such as heat therapy in conjunction with oral antifungals, and surgical interventions such as cryosurgery or surgical excision

Dokic, Yelena

Rosen, Theodore

Verstovsek, Gordana

English

DigitalCommons@The Texas Medical Center

The Texas Medical Center Library DigitalCommons@TMC Faculty and Staff Publications Baylor College of Medicine 12-1-2023 Chromoblastomycosis Presenting as a Solitary Lesion in a Non-endemic Region Yelena Dokic Gordana Verstovsek Theodore Rosen Follow this and additional works at: https://digitalcommons.library.tmc.edu/baylor_docs  Part of the Dermatology Commons, Infectious Disease Commons, Medical Sciences Commons, and the Skin and Connective Tissue Diseases Commons Review began 11/20/2023 Review ended 11/27/2023 Published 12/01/2023© Copyright 2023Dokic et al. This is an open access articledistributed under the terms of the CreativeCommons Attribution License CC-BY 4.0.,which permits unrestricted use, distribution,and reproduction in any medium, providedthe original author and source are credited.Chromoblastomycosis Presenting as a SolitaryLesion in a Non-endemic RegionYelena Dokic  , Gordana Verstovsek  , Theodore Rosen 1. Dermatology, Baylor College of Medicine, Houston, USA 2. Pathology and Immunology, Michael E. DeBakeyVeterans Affairs Medical Center, Baylor College of Medicine, Houston, USA 3. Dermatology, Michael E. DeBakeyVeterans Affairs Medical Center, Baylor College of Medicine, Houston, USACorresponding author: Yelena Dokic, yelena.dokic@bcm.eduAbstractChromoblastomycosis is a neglected tropical disease typically found in endemic tropical and subtropicalregions. Herein, we discuss a rare case of a 55-year-old man in Texas who presented with an exophyticpapule on the forearm, diagnosed to have chromoblastomycosis by shave biopsy and subsequenthistopathological analysis. Treatment options for chromoblastomycosis include long-term oral antifungaltherapy with itraconazole, physical modalities such as heat therapy in conjunction with oral antifungals,and surgical interventions such as cryosurgery or surgical excision.Categories: Dermatology, HIV/AIDS, Infectious DiseaseKeywords: copper penny spores, medlar bodies, cutaneous fungal infections, deep fungal infections, dermatology,infectious disease, itraconazole, dematiaceous fungus, chromoblastomycosisIntroductionChromoblastomycosis is a neglected tropical disease typically found in endemic tropical and subtropicalregions such as rural Latin America, the Caribbean, Africa, and Asia [1,2]. It is typically caused bytranscutaneous inoculation of the fungus after handling an environmental source, such as soil, treebranches, plant debris, and wood. It is rare for chromoblastomycosis to present in the United States,especially as a solitary papule, but such is the case for our patient. Herein, we present an unusual case ofchromoblastomycosis in a 55-year-old man presenting as a single lesion on a patient’s forearm in Texas, anon-endemic region.Case PresentationA 55-year-old African-American man with well-controlled HIV (most recent CD4 count was 740 cells/µL andmost recent viral load was <20 copies/mL detected) and Parkinson's disease presented with a 0.6 cm x 0.8cm exophytic papule on the forearm with a rolled border and central yellow and red crusting (Figure 1).1 2 3 Open Access CaseReport  DOI: 10.7759/cureus.49791How to cite this articleDokic Y, Verstovsek G, Rosen T (December 01, 2023) Chromoblastomycosis Presenting as a Solitary Lesion in a Non-endemic Region. Cureus15(12): e49791. DOI 10.7759/cureus.49791FIGURE 1: Exophytic papule on the right forearmThe lesion had been present for about one month prior to presentation and was slowly growing. The patientdenied any insect bites or previous skin cancers. He is a police officer by occupation and denies travelingoutside of Texas. The only traveling he had done was to East Texas two months before the lesion presentedon his arm. He stated that he was clearing tree branches and bushes at a house in East Texas just prior to thelesion appearing on his forearm and that he might have suffered penetrating trauma from working in thefoliage. The rest of the physical exam was unremarkable.The differential diagnosis for the lesion included basal cell carcinoma, squamous cell carcinoma, foreignbody granuloma, an infectious process, and other neoplastic or inflammatory processes. A shave biopsy ofthe papule on the right forearm was conducted, and histopathology revealed pyogranulomatousinflammation associated with pigmented fungal organisms (Figures 2, 3). Within the inflammation, therewere numerous brown-pigmented round yeast structures, some showing broad-based budding. The brownstructures, also known as Medlar bodies, are often likened to resembling copper pennies (Figure 4). A fungalGrocott-Gömöri's methenamine silver stain was conducted, which further highlighted the fungal organisms(Figure 5). The findings were deemed to be consistent with the presence of dematiaceous fungus, specificallychromoblastomycosis.2023 Dokic et al. Cureus 15(12): e49791. DOI 10.7759/cureus.49791 2 of 6FIGURE 2: Numerous dermal neutrophilic microabscesses (A)surrounded by epithelioid histiocytes (H) (20x)FIGURE 3: Darker staining microabscesses (arrow) with adjacent palergranuloma (arrowhead) (40x).2023 Dokic et al. Cureus 15(12): e49791. DOI 10.7759/cureus.49791 3 of 6FIGURE 4: Within the inflammation, Medlar bodies (arrows) are presentwithin giant cells and surrounded by neutrophils (200x)FIGURE 5: GMS stain highlighting the fungal organisms in black (200x)The patient was notified regarding the biopsy results and the likely association with his previous yard work.Although the entire lesion was ostensibly removed in the biopsy, he was prescribed itraconazole 200 mgdaily for two months as a prophylactic measure. It was also recommended that he start applying a heatingpad to the biopsy site three times daily for 10-15 minutes for two months, while on the itraconazole.2023 Dokic et al. Cureus 15(12): e49791. DOI 10.7759/cureus.49791 4 of 6DiscussionChromoblastomycosis is a granulomatous fungal infection of the skin and subcutaneous tissue that iscaused by dematiaceous fungi, primarily of the genera Fonsecaea, Phialophora, and Cladophialophora [1,2].Endemic to tropical and subtropical regions, it is rarely found in the United States.The exact pathogenesis of chromoblastomycosis can be via direct percutaneous inoculation, inhalation, andhematogenous dissemination. The fungus is found in soil, plants, and decaying wood. If organic materialthat is contaminated by the fungal organism is implicated in a traumatic injury, then percutaneousimplantation of the fungus may occur [3].Once inoculated into the skin, chromoblastomycosis is a locally progressive disease and can continue togrow over many years. Chromoblastomycosis can have a wide variety of morphologies [4]. It classicallypresents on the lower extremities of middle-aged men working in rural areas of tropical climates asverrucous reddish-purple papules. The lesions can be painless or mildly pruritic. Weeks to months later,similar lesions tend to appear peripherally, with the lesions growing centrifugally. Late lesions can appear asannular with central scarring. If the lesions are left to continue growing, entire limbs can become involved,covered with large, vegetative, verrucous plaques. Although the disease tends to primarily grow locally,satellite lesions can occur rarely, which are thought to spread via lymphatics.The diagnosis of chromoblastomycosis is confirmed by histopathological examination of skin biopsyspecimens, which typically reveal muriform fungal cells (Medlar bodies) within the granulomatous infiltrate.Fungal culture may also be helpful in identifying the causative fungal species.Therapeutic options for chromoblastomycosis center around oral antifungal agents, such as itraconazole,terbinafine, or posaconazole. Long-term itraconazole treatment (200-400 mg daily) is the first-line therapy,with a duration ranging from several months to even years, depending on the clinical severity [5].Terbinafine (250-500 mg daily) serves as a second-line treatment. Posaconazole (400 mg twice a day) may beconsidered for refractory cases, but its high cost limits its widespread use. Although fluconazole has one ofthe highest in vitro minimum fungicidal concentrations for the causative species of chromoblastomycosis, itmay be used at 300 mg weekly or 150 mg daily. Alternating weeks between daily itraconazole and dailyterbinafine therapy (itraconazole 200-400 mg/day and terbinafine 500-750 mg/day) is also an option [5].Heat therapy (local surface temperature of 46°C) is often used in conjunction with oral antifungals, as it hasproven effective in causing involution of the fungal lesions [6].Cryosurgery, involving the application of liquid nitrogen to freeze and destroy the affected tissue, can beused in combination with oral antifungals as a possible treatment for chromoblastomycosis [7]. This optionis more effective for smaller lesions, fewer lesions, and lesions that are not in anatomically sensitive areas. Itshould be avoided in areas with skin folds to prevent secondary fibrosis and scarring [7]. Counseling skin ofcolor patients regarding the possible side effects of hypopigmentation following cryotherapy is alsoimportant.Surgical excision with appropriate margins is recommended for small, early lesions to facilitate completeremoval of the fungal elements and prevent further spread. Curettage with electrodessication is generallynot advised, as it may promote the lymphatic spread of the infection [8]. Other potential destructive methodsinclude photodynamic therapy (PDT), CO2 laser photocoagulation, and immunoadjuvants such as topicalimiquimod [5].Despite the challenges in treating chromoblastomycosis, early diagnosis, and appropriate management canlead to significant improvement and even complete resolution of the disease.ConclusionsOur patient was a 55-year-old man in Texas who presented with an exophytic papule on the forearm and wasdiagnosed with biopsy-proven chromoblastomycosis. Several treatment modalities exist, and our patient iscurrently receiving itraconazole treatment with heating pads. Although rare, chromoblastomycosis canpresent as a solitary papule in the United States, a non-endemic region.Additional InformationAuthor ContributionsAll authors have reviewed the final version to be published and agreed to be accountable for all aspects of thework.Concept and design:  Yelena Dokic, Theodore Rosen, Gordana Verstovsek2023 Dokic et al. Cureus 15(12): e49791. DOI 10.7759/cureus.49791 5 of 6Acquisition, analysis, or interpretation of data:  Yelena Dokic, Theodore Rosen, Gordana VerstovsekDrafting of the manuscript:  Yelena Dokic, Theodore Rosen, Gordana VerstovsekCritical review of the manuscript for important intellectual content:  Yelena Dokic, Theodore Rosen,Gordana VerstovsekSupervision:  Yelena Dokic, Theodore Rosen, Gordana VerstovsekDisclosuresHuman subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: Incompliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/servicesinfo: All authors have declared that no financial support was received from any organization for thesubmitted work. Financial relationships: All authors have declared that they have no financialrelationships at present or within the previous three years with any organizations that might have aninterest in the submitted work. Other relationships: All authors have declared that there are no otherrelationships or activities that could appear to have influenced the submitted work.References1. Queiroz-Telles F, Esterre P, Perez-Blanco M, Vitale RG, Salgado CG, Bonifaz A: Chromoblastomycosis: anoverview of clinical manifestations, diagnosis and treatment. Med Mycol. 2009, 47:3-15.10.1080/136937808025380012. Queiroz-Telles F, de Hoog S, Santos DW, et al.: Chromoblastomycosis. Clin Microbiol Rev. 2017, 30:233-76.10.1128/CMR.00032-163. Rubin HA, Bruce S, Rosen T, McBride ME: Evidence for percutaneous inoculation as the mode oftransmission for chromoblastomycosis. J Am Acad Dermatol. 1991, 25:951-954. 10.1016/0190-9622(91)70292-a4. Mugleston BJ, Usatine RP, Rosen T: Wide morphologic variability of chromoblastomycosis in the WesternHemisphere. Skinmed. 2016, 14:423-7.5. Asad U, Hopkins C, Rosen T: Deep Fungal Infections. Fitzpatrick's Therapeutics: A Clinician's Guide toDermatologic Treatment. Kang S, Goh CL, Kim N, Kwatra S (ed): McGraw-Hill, New York; 2023. 613-27.6. Tagami H, Ohi M, Aoshima T, Moriguchi M, Suzuki N, Yamada M: Topical heat therapy for cutaneouschromomycosis. Arch Dermatol. 1979, 115:740-1. 10.1001/archderm.115.6.7407. Rosen T, Bonifaz A, Fierro-Arias L, Peniche-Castellanos A, Vázquez-González D: Chromoblastomycosis.Dermatological Cryosurgery and Cryotherapy. Abramovits W, Graham G, Har-Shai Y, Strumia R (ed):Springer, London; 2016.8. Bonifaz A, Paredes-Solis V, Saul A: Treating chromoblastomycosis with systemic antifungals . Expert OpinPharmacother. 2004, 5:247-54. 10.1517/14656566.5.2.2472023 Dokic et al. Cureus 15(12): e49791. DOI 10.7759/cureus.49791 6 of 6

Chromoblastomycosis Presenting as a Solitary Lesion in a Non-endemic Region

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Chromoblastomycosis Presenting as a Solitary Lesion in a Non-endemic Region

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