Tropical/Infectoparasitary Dermatology
Disseminated mucocutaneous trichosporonosis in a patient with histiocytic sarcoma
Arturo Robles-Tenorioa, Rossy Anahí Rivas-Lópezb, Alexandro Bonifazc, Víctor Manuel Tarango-Martíneza,
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a Instituto Dermatológico de Jalisco “Dr. José Barba Rubio”, Zapopan, Jalisco, Mexico
b Centro Dermatológico de Sinaloa "Dr. Jesús Rodolfo Acedo Cárdenas", Culiacán, Sinaloa, México
c Servicio de dermatología y departamento de Micología, Hospital General de México “Dr. Eduardo Liceaga”, Ciudad de México, México
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array:1 [ 0 => array:4 [ "autoresLista" => "Arturo Robles-Tenorio, Rossy Anahí Rivas-López, Alexandro Bonifaz, Víctor Manuel Tarango-Martínez" "autores" => array:4 [ 0 => array:3 [ "nombre" => "Arturo" "apellidos" => "Robles-Tenorio" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "Rossy Anahí" "apellidos" => "Rivas-López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Alexandro" "apellidos" => "Bonifaz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:4 [ "nombre" => "Víctor Manuel" "apellidos" => "Tarango-Martínez" "email" => array:1 [ 0 => "drtarangovic@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Instituto Dermatológico de Jalisco “Dr. José Barba Rubio”, Zapopan, Jalisco, Mexico" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Centro Dermatológico de Sinaloa "Dr. Jesús Rodolfo Acedo Cárdenas", Culiacán, Sinaloa, México" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de dermatología y departamento de Micología, Hospital General de México “Dr. Eduardo Liceaga”, Ciudad de México, México" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 905 "Ancho" => 905 "Tamanyo" => 194055 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Large, necrotic face ulcers affecting the cheek and the oral mucosa.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case report</span><p id="par0005" class="elsevierStylePara elsevierViewall">A previously healthy 57-year-old indigenous woman presented to the clinic with an 18-month history of facial wounds, progressive pleuritic pain, dyspnea, productive cough, weight loss, fever, and fatigue. On examination, there were multiple necrotic ulcers on the left cheek, nose, and lips (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Submaxillary, cervical, and axillary lymph nodes appeared swollen. Laboratory results showed anemia (4.6 g/dL) and LDH elevation (1477 UI/L). Serologies for HIV 1/2, Hepatitis B and C were negative. Computed tomography exhibited basal, bilateral consolidations and atelectasis, hepatomegaly, and free intraperitoneal fluid. Face ulcer smear and histopathology reported budding yeast cells and blastoconidia (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Despite receiving liposomal amphotericin B, the patient presented lesions dissemination to the oral cavity, lower gastrointestinal bleeding, mixed shock, and died fifteen days after admission. Face ulcer culture (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>) and axillary lymph node histopathology were collected post-mortem. <span class="elsevierStyleItalic">Trichosporon asahii</span> was identified by matrix-assisted laser desorption ionization-time of flight mass spectrometry (MALDI-TOF MS, Vitek-MS®). Axillary node histopathology showed Histiocytic Sarcoma (HS), with positive CD68 and CD163 on immunohistochemistry.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Discussion</span><p id="par0010" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Trichosporon</span> spp. are found as commensal and pathogenic fungi, capable of evading the immune response and generating antimicrobial resistance through biofilm formation, metabolic, and phenotypic features.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a><span class="elsevierStyleItalic">Trichosporon asahii</span> affects the skin, mucous membranes, and internal viscera of patients with immunosuppression, hematolymphoid malignancies, or chemotherapy-induced neutropenia.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Disseminated mucocutaneous trichosporonosis presents as rapidly progressive papulonodular lesions and necrotic ulcers, similar to cryptococcosis.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Trichosporonosis is suspected by observing hyphae, arthroconidia, and blastoconidia on a smear and histopathology.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The definitive diagnosis relies on species identification from culture colonies by chromogenic or biochemical methods.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a> MALDI-TOF MS might soon replace these processes, providing higher accuracy yields.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Other techniques such as flow cytometry, polymerase chain reaction, and gene sequencing are still under investigation.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Comparative evidence of antifungal therapy is limited. In a systematic review of 203 cases, voriconazole had the highest favorable outcome rate (73.6%) in patients with hematological neoplasms, and also the best <span class="elsevierStyleItalic">in vitro</span> activity against <span class="elsevierStyleItalic">Trichosporon spp.</span> Similarly, in Mexico, minimum inhibitory concentrations were the lowest for triazoles, higher for amphotericin B, and the highest for echinocandins.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Together, these results are in line with treatment guidelines, where voriconazole is the preferred antifungal. In the present case study, the authors used liposomal amphotericin B due to availability at the authors’ institution.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Therapeutic success depends on the underlying disease status. Here, the patient had an advanced case of HS. This rare and aggressive cancer represents 1% of all hematolymphoid neoplasms.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> HS usually affects lymph nodes, but it can speedily disseminate to several organs. On histopathology, the tumor shows diffuse proliferation of neoplastic cells with eosinophilic cytoplasm and eccentric nuclei. Immunohistochemistry is positive for CD163, CD68, and lysozyme. Epithelial, melanocytic, myeloid, Langerhans, B, and T cell markers are negative. As in this patient, HS is often a late diagnosis and has an estimated survival of fewer than two years.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In conclusion, trichosporonosis and HS are infrequent, clinically challenging diseases. A swift, interdisciplinary action between dermatology, oncology, and infectious diseases specialists is of utmost importance.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Authors’ contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Arturo Robles-Tenorio: Critically reviewed the literature, analyzed the data, and wrote the final version of the manuscript.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Rossy Anahí Rivas-López: Prepared the original draft, was responsible of study conception and data collection.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Alexandro Bonifaz: Critically reviewed the manuscript, provided supporting references, and approved the final version of the manuscript.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Víctor Manuel Tarango-Martínez: Prepared the original draft, collected the data, and approved the final version of the manuscript.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:3 [ "identificador" => "xres1581937" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1423930" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 3 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 4 => array:2 [ "identificador" => "sec0015" "titulo" => "Financial support" ] 5 => array:2 [ "identificador" => "sec0020" "titulo" => "Authors’ contributions" ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-10-25" "fechaAceptado" => "2021-01-15" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1423930" "palabras" => array:5 [ 0 => "Hematologic neoplasms" 1 => "Histiocytic sarcoma" 2 => "Trichosporon" 3 => "Trichosporonosis" 4 => "Ulcer" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Trichosporon asahii</span> is the causal agent of trichosporonosis. Patients with immunosuppression or hematological malignancies are at higher risk of infection. Skin and mucosal involvement appear as fast-growing papulonodular lesions and necrotic ulcers. Internal organ dissemination is lethal. Therapeutic success depends on the underlying disease. Here, the authors present the first case of disseminated mucocutaneous trichosporonosis in a patient with a post-mortem diagnosis of histiocytic sarcoma, a rare and aggressive haematolymphoid neoplasm. Regretfully, death occurred despite treatment with liposomal amphotericin B and supportive measures, showcasing the fatality of both diseases.</p></span>" ] ] "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Robles-Tenorio A, Rivas-López RA, Bonifaz A, Tarango-Martínez VM. Disseminated mucocutaneous trichosporonosis in a patient with histiocytic sarcoma. An Bras Dermatol. 2021;96:595–597.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital General de Occidente, Zapopan, México.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 905 "Ancho" => 905 "Tamanyo" => 194055 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Large, necrotic face ulcers affecting the cheek and the oral mucosa.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 493 "Ancho" => 1305 "Tamanyo" => 123185 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Face ulcer examinations. (A), Budding yeast cells and (B), PAS stained blastoconidia are observed on a Gram smear and on histopathology, respectively.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 490 "Ancho" => 1305 "Tamanyo" => 93440 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Face ulcer culture. (A), <span class="elsevierStyleItalic">Trichosporon spp.</span> colonies showed a raised, waxy appearance with radial furrows on Sabouraud culture. 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