Case Report
Unsuspected Visceral Leishmaniasis Infiltrating a Squamous Cell Carcinoma
Leishmaniasis visceral insospechada infiltrando un carcinoma epidermoide
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Armengot-Carbó, R. Carmena-Ramón, B. Rodrigo-Nicolás, J. Ferrando-Marco" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Armengot-Carbó" "email" => array:1 [ 0 => "miquelarmengot@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "R." "apellidos" => "Carmena-Ramón" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "B." "apellidos" => "Rodrigo-Nicolás" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Ferrando-Marco" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Arnau de Vilanova, Valencia, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Arnau de Vilanova, Valencia, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Leishmaniasis visceral insospechada infiltrando un carcinoma epidermoide" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 720 "Ancho" => 960 "Tamanyo" => 245390 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Extensive, predominantly lymphocytic inflammatory infiltrate and abundant macrophages; round or oval basophilic structures can be seen both inside the cytoplasm (arrow) and in the intercellular matrix (arrowhead), consistent with <span class="elsevierStyleItalic">Leishmania</span> amastigotes (hematoxylin-eosin, original magnification ×400).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Amastigotes of the genus <span class="elsevierStyleItalic">Leishmania</span> have been observed in biopsies of apparently unrelated lesions in patients who have both AIDS and visceral leishmaniasis.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> We report the case of a man in whom the presence of the parasite was detected in a perianal squamous cell carcinoma. This association has not been reported in the literature to date.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case Description</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 40-year-old human immunodeficiency virus (HIV)-infected homosexual man had been receiving antiretroviral therapy (abacavir, etravirine, and raltegravir) since 2005. He had been referred to our department on several occasions for evaluation of perianal condylomata, which were treated with cryotherapy. In April 2010, he was again referred for an erosive, erythematous, verrucous perianal plaque that had developed several months earlier (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A biopsy was performed to rule out malignant transformation.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histopathology revealed marked epidermal acanthosis, pleomorphic keratinocytes, and the presence of atypical mitotic figures, confirming the suspected clinical diagnosis of squamous cell carcinoma (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). In the dermis, numerous amastigotes of the genus <span class="elsevierStyleItalic">Leishmania</span> were observed both inside the macrophages and in the intercellular matrix (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). In situ hybridization for high-risk human papillomavirus (HPV) revealed strong, punctate, nuclear staining. Immunohistochemistry was positive for p16 in neoplastic areas.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">On elicitation of additional history, the patient reported that his dog had died of leishmaniasis 4 years earlier and that he frequently traveled from his home in Valencia to Morocco on business.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Blood tests revealed a CD4 cell count of 90/μL, an undetectable HIV load (<20 copies/mL), and an erythrocyte sedimentation rate of 116<span class="elsevierStyleHsp" style=""></span>mm/h. The total white blood cell count and platelet count were normal and the patient was only slightly anemic (hemoglobin 11.9<span class="elsevierStyleHsp" style=""></span>g/dL).</p><p id="par0030" class="elsevierStylePara elsevierViewall">The patient refused to undergo bone marrow sampling, so the infectious diseases department started empiric treatment with meglumine antimoniate (20<span class="elsevierStyleHsp" style=""></span>mg/kg of a pentavalent antimony compound once daily for 28 days). The patient was also scheduled for excision of the tumor. Histologic examination of the excised tissue confirmed the presence of <span class="elsevierStyleItalic">Leishmania</span> organisms, despite completion of the antimonial treatment. The patient then agreed to a bone marrow procedure; involvement of the marrow was detected, confirming the diagnosis of visceral leishmaniasis. In view of these findings, the patient was prescribed treatment with liposomal amphotericin B. Because his CD4 cell count remained below 200/μL, on completion of treatment he began secondary chemoprophylaxis with monthly administration of liposomal amphotericin B. After 9 months, no clinical or analytical signs of visceral leishmaniasis were evident. He currently attends regular follow-up visits at the infectious diseases department.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">Leishmaniasis is caused by infection with protozoa of the genus <span class="elsevierStyleItalic">Leishmania</span>, which are spread by female sandflies of the genera <span class="elsevierStyleItalic">Phlebotomus</span> and <span class="elsevierStyleItalic">Lutzomyia</span>. The <span class="elsevierStyleItalic">Leishmania</span> protozoa parasitize the monocyte-macrophage system in the form of amastigotes. In Spain, dogs are the main reservoir of the causative species,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a><span class="elsevierStyleItalic">Leishmania infantum.</span><a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Immunocompromised patients bitten by an infected vector are at high risk of developing visceral leishmaniasis.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Most cases of visceral leishmaniasis were once found in children, but at present 70% occur in adults, and 50% of such cases are in HIV-positive individuals.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> Spain and the rest of southwestern Europe have the world's highest prevalence of coinfection, and intravenous drug users are at the greatest risk.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1-6</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">HIV/<span class="elsevierStyleItalic">Leishmania</span> organism coinfection favors the progression of both infections. CD4+ type 1 helper T cells, which are depleted in HIV-infected patients, play a fundamental role in the immune response to the parasite. HIV infection therefore increases the risk of developing visceral leishmaniasis by a factor of 100 to 2300, worsens prognosis, and decreases the likelihood of therapeutic response.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,6</span></a> Furthermore, the presence of <span class="elsevierStyleItalic">Leishmania</span> amastigotes has been shown to increase HIV replication, diminish immune status, and hasten progression to AIDS.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Visceral leishmaniasis spreads to the skin more frequently in HIV-infected patients than in immunocompetent patients<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and manifests as lesions spanning a wide clinical spectrum.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,7</span></a> In addition, the presence of <span class="elsevierStyleItalic">Leishmania</span> organisms has been observed in biopsy specimens from patients with skin conditions not caused by the parasite, such as Kaposi sarcoma, dermatofibromas, psoriasis, aphthous ulcers, herpes simplex, herpes zoster, bacillary angiomatosis, tattoos, and rheumatoid nodules.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">In our patient, the disease was detected incidentally on biopsy of a perianal squamous cell carcinoma. To our knowledge, this association has not been previously reported. The patient's epithelioma does not appear to have been caused by the parasite; had it been, it would suggest a relationship with chronic HPV infection. The growth was probably caused by massive dissemination of the parasite in this severely immunocompromised patient. Local inflammation caused by growth of the carcinoma, accompanied by an abundance of histiocytes in a heavily parasitized mononuclear phagocyte system, would explain findings of abundant amastigotes.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,8</span></a> This finding in our patient led to further tests though which previously unsuspected visceral involvement was detected. Appropriate systemic treatment, as described elsewhere,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> was then prescribed.</p><p id="par0060" class="elsevierStylePara elsevierViewall">This is the first report of an association between <span class="elsevierStyleItalic">Leishmania</span> parasites and squamous cell carcinoma. In HIV-positive patients in whom skin biopsy reveals evidence of <span class="elsevierStyleItalic">Leishmania</span> parasites, underlying visceral leishmaniasis should be suspected. Bone marrow sampling would seem to be indicated, as the results will guide treatment choice and ultimately influence prognosis.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflicts of Interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres95848" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec83008" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres95849" "titulo" => "Resumen" ] 3 => array:2 [ "identificador" => "xpalclavsec83007" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case Description" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflicts of Interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec83008" "palabras" => array:4 [ 0 => "Squamous cell carcinoma" 1 => "Leishmaniasis" 2 => "AIDS" 3 => "Human immunodeficiency virus" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec83007" "palabras" => array:4 [ 0 => "Carcinoma epidermoide" 1 => "Leishmaniasis" 2 => "Sida" 3 => "VIH" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Amastigotes of the genus <span class="elsevierStyleItalic">Leishmania</span> have been observed in biopsies of apparently unrelated lesions in patients with AIDS and visceral leishmaniasis. We describe the case of a 40-year-old man with human immunodeficiency virus infection and severe immunodepression in whom the presence of the parasite was detected as an incidental finding on histological study of a perianal squamous cell carcinoma. This finding led to the diagnosis and subsequent treatment of previously unsuspected visceral leishmaniasis. In a review of the literature we have found no previous examples of this association.</p>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">En pacientes con SIDA y leishmaniasis visceral ha sido descrita la presencia de amastigotes de <span class="elsevierStyleItalic">Leishmania</span> en biopsias realizadas para estudiar diversas lesiones con las que no guardan aparente relación causal. Presentamos el caso de un varón de 40 años, VIH positivo severamente inmunodeprimido, en el que se observó incidentalmente la presencia del parásito al estudiar histológicamente un carcinoma epidermoide perianal. Dicho hallazgo permitió el diagnóstico y tratamiento de una leishmaniasis visceral insospechada. No hemos encontrado en la literatura ejemplos previos de esta asociación.</p>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Armengot-Carbó M, et al. Leishmaniasis visceral insospechada infiltrando un carcinoma epidermoide. Actas Dermosifiliogr.2012;103:321-323.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 713 "Ancho" => 950 "Tamanyo" => 159010 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Erosive, erythematous, verrucous plaque in the perianal region, several months after initial appearance.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 720 "Ancho" => 960 "Tamanyo" => 276114 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Epidermis with marked acanthosis, epithelial immaturity, cell pleomorphism, and atypical mitotic figures in various strata (hematoxylin-eosin, original magnification ×40; inset ×200).</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 720 "Ancho" => 960 "Tamanyo" => 245390 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Extensive, predominantly lymphocytic inflammatory infiltrate and abundant macrophages; round or oval basophilic structures can be seen both inside the cytoplasm (arrow) and in the intercellular matrix (arrowhead), consistent with <span class="elsevierStyleItalic">Leishmania</span> amastigotes (hematoxylin-eosin, original magnification ×400).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Leishmaniasis and rheumatoid nodulosis in a patient with HIV infection" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "I. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 7 | 9 | 16 |
| 2024 Octubre | 56 | 37 | 93 |
| 2024 Septiembre | 69 | 17 | 86 |
| 2024 Agosto | 91 | 54 | 145 |
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| 2023 Agosto | 45 | 14 | 59 |
| 2023 Julio | 60 | 35 | 95 |
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| 2023 Marzo | 30 | 30 | 60 |
| 2023 Febrero | 54 | 22 | 76 |
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| 2022 Septiembre | 21 | 41 | 62 |
| 2022 Agosto | 26 | 41 | 67 |
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| 2022 Febrero | 28 | 24 | 52 |
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| 2021 Diciembre | 30 | 35 | 65 |
| 2021 Noviembre | 40 | 46 | 86 |
| 2021 Octubre | 47 | 62 | 109 |
| 2021 Septiembre | 29 | 36 | 65 |
| 2021 Agosto | 29 | 31 | 60 |
| 2021 Julio | 35 | 24 | 59 |
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| 2020 Septiembre | 28 | 14 | 42 |
| 2020 Agosto | 20 | 21 | 41 |
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| 2020 Junio | 37 | 26 | 63 |
| 2020 Mayo | 23 | 16 | 39 |
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| 2020 Marzo | 29 | 21 | 50 |
| 2020 Febrero | 2 | 1 | 3 |
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| 2019 Noviembre | 4 | 0 | 4 |
| 2019 Octubre | 0 | 1 | 1 |
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| 2018 Marzo | 1 | 0 | 1 |
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| 2017 Diciembre | 76 | 6 | 82 |
| 2017 Noviembre | 60 | 4 | 64 |
| 2017 Octubre | 50 | 4 | 54 |
| 2017 Septiembre | 56 | 10 | 66 |
| 2017 Agosto | 69 | 9 | 78 |
| 2017 Julio | 54 | 7 | 61 |
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| 2017 Febrero | 44 | 4 | 48 |
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| 2016 Noviembre | 83 | 7 | 90 |
| 2016 Octubre | 91 | 8 | 99 |
| 2016 Septiembre | 163 | 4 | 167 |
| 2016 Agosto | 76 | 7 | 83 |
| 2016 Julio | 39 | 8 | 47 |
| 2016 Junio | 10 | 0 | 10 |
| 2016 Mayo | 8 | 3 | 11 |
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| 2016 Marzo | 10 | 0 | 10 |
| 2016 Febrero | 9 | 2 | 11 |
| 2016 Enero | 5 | 2 | 7 |
| 2015 Diciembre | 7 | 0 | 7 |
| 2015 Noviembre | 24 | 6 | 30 |
| 2015 Octubre | 19 | 4 | 23 |
| 2015 Septiembre | 8 | 2 | 10 |
| 2015 Agosto | 13 | 1 | 14 |
| 2015 Julio | 174 | 7 | 181 |
| 2015 Junio | 126 | 6 | 132 |
| 2015 Mayo | 172 | 17 | 189 |
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| 2015 Enero | 129 | 25 | 154 |
| 2014 Diciembre | 131 | 6 | 137 |
| 2014 Noviembre | 106 | 17 | 123 |
| 2014 Octubre | 154 | 19 | 173 |
| 2014 Septiembre | 114 | 11 | 125 |
| 2014 Agosto | 138 | 19 | 157 |
| 2014 Julio | 151 | 25 | 176 |
| 2014 Junio | 168 | 17 | 185 |
| 2014 Mayo | 178 | 16 | 194 |
| 2014 Abril | 109 | 11 | 120 |
| 2014 Marzo | 130 | 9 | 139 |
| 2014 Febrero | 96 | 9 | 105 |
| 2014 Enero | 79 | 5 | 84 |
| 2013 Diciembre | 78 | 4 | 82 |
| 2013 Noviembre | 66 | 14 | 80 |
| 2013 Octubre | 55 | 4 | 59 |
| 2013 Septiembre | 67 | 3 | 70 |
| 2013 Agosto | 51 | 11 | 62 |
| 2013 Julio | 16 | 11 | 27 |
| 2013 Junio | 10 | 20 | 30 |
| 2013 Mayo | 14 | 11 | 25 |
| 2013 Abril | 11 | 20 | 31 |
| 2013 Marzo | 21 | 11 | 32 |
| 2013 Febrero | 31 | 3 | 34 |
| 2013 Enero | 103 | 5 | 108 |
| 2012 Diciembre | 56 | 5 | 61 |
| 2012 Agosto | 0 | 1 | 1 |
| 2012 Julio | 2 | 0 | 2 |
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