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Mycosis fungoides-like lesions in a patient with diffuse cutaneous sporotrichosis
Lesiones que semejan una micosis fungoide en un paciente con esporotricosis cutánea difusa
Adriana Cotino Sáncheza, Bertha Torres-Alvarezb, Teodoro Gurrola Moralesa, Silvia Méndez Martíneza, Mauricio Saucedo Gáratea, Juan Pablo Castanedo-Cazaresb,
Corresponding author
castanju@yahoo.com

Corresponding author.
a Department of Pathology, Hospital General de Durango, Universidad Juárez del Estado de Durango, Durango, Mexico
b Department of Dermatology, Hospital Central Dr. Ignacio Morones Prieto, Universidad Autónoma de San Luis Potosí, San Luis Potosí, Mexico
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sporotrichosis is a worldwide fungal infection caused by the inhalation or traumatic inoculation of spores of the dimorphic fungus <span class="elsevierStyleItalic">Sporothrix schenckii</span> complex&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;9&#44;19</span></a> It is a polymorphic disease that has two clinical forms&#44; localized and systemic &#40;disseminated&#41;&#44; although the most common presentation is a subacute or chronic subcutaneous process&#46; Molecular studies have shown that the <span class="elsevierStyleItalic">S&#46; schenckii</span> complex includes species that have not been completely characterized&#44; such as <span class="elsevierStyleItalic">Sporothrix albican</span>s&#44; <span class="elsevierStyleItalic">Sporothrix brasiliensis</span>&#44; <span class="elsevierStyleItalic">Sporothrix globosa</span>&#44; <span class="elsevierStyleItalic">Sporothrix luriei</span>&#44; <span class="elsevierStyleItalic">Sporothrix mexicana</span>&#44; and <span class="elsevierStyleItalic">S&#46; schenckii</span>&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Sporotrichosis has a relatively high incidence in Latin American countries such as Mexico&#44; where it is frequent in the central and occidental areas of the country&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#44;19</span></a> It is seen in both genders and at all ages&#44; although it is more common among children and young adults in this part of the world&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;6&#44;19</span></a> It is considered an occupational infectious disease because miners&#44; farmers&#44; gardeners&#44; and carpenters are frequently affected due to exposure to the natural habitat of this microorganism&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;6&#44;19</span></a> Indirect vectors such as rats&#44; mice&#44; cats&#44; and squirrels have also been described&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> We present the case of an alcoholic and iatrogenically immunosuppressed patient with disseminated cutaneous sporotrichosis with mycosis fungoides-like lesions&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 68-year-old male farmer from Durango&#44; Mexico&#44; presented to his local family physician with a 4-year history of asymptomatic scaly nodules and plaques on his face&#46; These lesions were treated with topical antibiotics and steroids with transient and partial involution&#46; Five months later&#44; the nodules and plaques progressively increased in size evolving into tumors&#46; Similar lesions appeared on the thorax&#44; abdomen and limbs&#46; Symptoms of malaise&#44; intermittent fever&#44; and a 10&#37; loss of his usual weight were also present&#46; The patient was reexamined by an internist who prescribed oral prednisone 50<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#44; and topical mometasone 0&#46;1&#37; with a presumptive diagnosis of mycosis fungoides&#44; supported by the clinical presentation and the histological study of a facial lesion&#46; After three weeks of treatment&#44; the patient exhibited clinical worsening with new nodular lesions&#44; ulceration&#44; and tumor enlargement&#46; The patient was referred to a dermatology center&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient had a 20-year history of high blood pressure with irregular treatment&#44; a 18 pack-year history of smoking&#44; and 500<span class="elsevierStyleHsp" style=""></span>g of alcohol consumption per week&#46; He did not remember any scratching&#44; trauma&#44; or skin puncture by thorns or organic material&#46; Physical examination revealed asymptomatic multiple scaly nodules&#44; tumors and ill-defined plaques 2 to 8-cm in diameter without tenderness&#44; disseminated to the head&#44; thorax and limbs&#44; some with ulceration &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Painless bilateral cervical and clavicular adenopathy was found&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A complete blood cell count showed hemoglobin of 9&#46;5<span class="elsevierStyleHsp" style=""></span>g&#47;dl&#44; and hematocrit of 29&#46;5&#37;&#46; The white cell count was 17&#44;769 neutrophils&#47;mm<span class="elsevierStyleSup">3</span>&#44; 2160 lymphocytes&#47;mm<span class="elsevierStyleSup">3</span>&#44; and 4080 eosinophils&#47;mm<span class="elsevierStyleSup">3</span>&#46; A buffy coat smear for S&#233;zary cells&#44; a chest X-ray and a thoracoabdominal CT scan were normal&#46; Serology for HIV was negative&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histologic examination of nodules on the right cheek and upper thorax revealed a suppurative granulomatous dermatitis&#46; A biopsy specimen showed hyperkeratosis&#44; acanthosis&#44; and a diffuse infiltrate of granulocytes&#44; lymphocytes&#44; and plasma cells throughout the dermis&#46; Periodic acid-Schiff &#40;PAS&#41; stain revealed the presence of yeast forms&#44; 2&#8211;4<span class="elsevierStyleHsp" style=""></span>mm with enhancement of their walls &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Special stains for bacteria and acid-fast bacilli were negative&#46; Tissue fungal culture on Sabouraud agar grew <span class="elsevierStyleItalic">Sporothrix schenckii</span>&#46; Septate hyphae with conidia arranged in rosette-like clusters were observed after staining with lactophenol aniline blue &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; The treatment was started with oral itraconazole&#44; 200<span class="elsevierStyleHsp" style=""></span>mg&#47;d which showed marked improvement over the first six weeks with normalization of white blood cells parameters&#44; and complete remission after 4 months of treatment &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Sporotrichosis is a polymorphic disease with lymphocutaneous&#44; fixed&#44; and disseminated clinical variants&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;9&#44;19</span></a> The most common lesions are fixed and lymphocutaneous&#46; The typical course of disease begins days to weeks after inoculation with a nodule or ulcer at the site of entrance&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;9&#44;19</span></a> Lesions enlarge slowly&#44; and form satellite nodules progressing along regional lymphatics&#46; Disseminated cutaneous sporotrichosis is characterized by more than three skin lesions in two different anatomical sites without extracutaneous involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">19</span></a> This clinical presentation is rare&#44; accounting for less than 2&#37; of the cases&#44; and is usually associated to immunosuppression&#44; where the fungus acts as an opportunistic pathogen&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;9&#44;19</span></a> Host immunity is hypoergic or anergic&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;5&#44;19</span></a> Risk factors include alcoholism&#44; diabetes&#44; AIDS&#44; pregnancy&#44; malnutrition&#44; malignancy&#44; and immunosuppressive therapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;19</span></a> In these settings&#44; lesions are usually nodules&#44; gums&#44; and ulcers&#46; Some disseminated variants have been associated to pyoverrucoid syndromes consisting of necrotic ulcers&#44; erythematous nodules&#44; and verrucous plaques with hematic crusts&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> However&#44; most cases are not easily recognized due to unusual histological and clinical manifestations for even years before the diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> These atypical presentations may be related to the pathogenicity&#44; virulence and infectivity of the <span class="elsevierStyleItalic">Sporothrix</span> strains&#44; geographical factors&#44; site of inoculation&#44; and immunological status of the patient&#44; among others&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;8</span></a> However&#44; no differences between the pathogenicity and clinical setting among several species of <span class="elsevierStyleItalic">Sporothrix</span> complex have been proved&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Although the diagnosis of cutaneous sporotrichosis is basically clinical&#44; it must be confirmed by culture&#46; Few conditions are considered in the differential diagnosis and these include leishmaniasis&#44; chromoblastomycosis&#44; cutaneous tuberculosis&#44; and chronic staphylococcal lymphangitis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">There are worldwide reports from endemic countries concerning infrequent cutaneous presentations&#46; Some include keratoacanthoma&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> sarcoidosis&#44;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">20</span></a> arachnidism&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> chronic ulcer&#44;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> cryptococcosis&#44;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">16</span></a> mycetoma&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">17</span></a> moluscum&#44;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> soft tissue tumor&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> leprosy&#44;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">18</span></a> and pyoderma gangrenosum&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We could not find previous reports of disseminated sporotrichosis manifesting as nodules and tumors such as in mycosis fungoides&#46; The similarity between these lesions and those observed in the tumoral stage of a cutaneous lymphoma may confuse the unskilled physician&#46; However&#44; the clinical history and histological features rule out this diagnosis&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">It is assumed that the <span class="elsevierStyleItalic">Sporothrix</span> cell-wall polysaccharide fraction is responsible for its immunogenicity&#46; After fungal antigens are processed and recognized&#44; a TH1 response is elicited&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> Interestingly&#44; we found suppurative granulomas in these lesions&#44; which are common among variants with preserved immunity&#44; such as fixed and lymphangitic&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Therefore&#44; we think that this clinical expression resembling mycosis fungoides-lesions could have been favored by an indeterminate inflammatory response&#46; This is supported by the absence of the expected tuberculoid granuloma observed in disseminated variants&#44; along with the chronic clinical setting&#44; and the parasitic fungal yeasts observed in tissues&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> Unfortunately&#44; the sporotrichin test was unavailable&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Concerning treatment&#44; a favorable outcome as that observed in this patient confirms the usefulness of itraconazole in sporotrichosis associated to immunosuppression and disseminated clinical forms&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;6</span></a> Although this case was complex&#44; it clearly demonstrates that clinical expression is greatly determined by the immune response to the fungus&#46; Therefore&#44; careful attention to the individual&#39;s personal life such as this patient&#39;s occupation&#44; his history of alcoholism and the fact that he lives in an endemic area&#44; as well as a thoughtful interpretation of the skin biopsy and appropriate culture&#44; should provide a correct diagnosis in similar cases&#46;</p></span></span>"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Sporotrichosis is a subacute or chronic mycosis acquired by traumatic inoculation or inhalation of fungal conidia&#46; It is caused by the dimorphic fungus <span class="elsevierStyleItalic">Sporothrix</span>&#44; which causes different clinical presentations&#44; being the cutaneous and lymphocutaneous variants being the most frequent&#46; The disseminated cutaneous form is a rare presentation occurring in a minority of cases in Mexico&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case report</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We report an atypical case of disseminated sporotrichosis in an alcoholic and iatrogenically immunosuppressed patient&#44; whose clinical lesions resembled tumoral-stage mycosis fungoides&#46; Histological examination and culture revealed the presence of <span class="elsevierStyleItalic">Sporothrix schenckii</span>&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conclusions</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The patient was treated with itraconazole 200<span class="elsevierStyleHsp" style=""></span>mg per day for 4 months with clinical resolution&#46; To the best of our knowledge&#44; this is the first report of this type of clinical manifestation&#46;</p></span>"
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        "resumen" => "<span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Antecedentes</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La esporotricosis es una micosis subaguda o cr&#243;nica adquirida por inoculaci&#243;n traum&#225;tica o inhalaci&#243;n de conidias f&#250;ngicas&#46; Est&#225; causada por el hongo dim&#243;rfico <span class="elsevierStyleItalic">Sporothrix</span>&#44; y puede presentar distintas manifestaciones cl&#237;nicas&#44; si bien las variantes linfocut&#225;nea y cut&#225;nea son las m&#225;s frecuentes&#46; La forma cut&#225;nea diseminada es una presentaci&#243;n infrecuente que se ha observado en una minor&#237;a de casos en M&#233;xico&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Caso cl&#237;nico</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Informamos de un caso at&#237;pico de esporotricosis diseminada en un paciente alcoh&#243;lico y con inmunosupresi&#243;n iatrog&#233;nica&#44; cuyas lesiones se asemejaban a una micosis fungoide en fase tumoral&#46; La observaci&#243;n histol&#243;gica y el cultivo demostraron la presencia de <span class="elsevierStyleItalic">Sporothrix schenckii</span>&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusiones</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">El paciente fue tratado con 200<span class="elsevierStyleHsp" style=""></span>mg de itraconazol diarios durante 4 meses con resoluci&#243;n cl&#237;nica&#46; Este es el primer informe en relaci&#243;n con esta manifestaci&#243;n cl&#237;nica&#46;</p></span>"
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          "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">Suppurative granuloma with central microabscesses composed of neutrophils surrounded by epithelioid cells&#44; and a peripheral zone infiltrated by lymphoid and plasma cells &#40;hematoxylin-eosin stain&#44; 100&#215;&#41; &#40;left&#41;&#46; An oval body &#40;4<span class="elsevierStyleHsp" style=""></span>&#956;m in diameter&#41; with a lighter center and darker stain at the periphery is seen&#44; suggestive of <span class="elsevierStyleItalic">Sporothrix</span> yeast &#40;PAS stain&#44; 400&#215;&#41; &#40;right&#41;&#46;</p>"
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                      "titulo" => "<span class="elsevierStyleItalic">Sporothrix schenckii</span> and sporotrichosis"
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                        0 => array:2 [
                          "etal" => false
                          "autores" => array:3 [
                            0 => "M&#46;B&#46; Barros"
                            1 => "R&#46; de Almeida Paes"
                            2 => "A&#46;O&#46; Schubach"
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                        "fecha" => "2011"
                        "volumen" => "24"
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                0 => array:2 [
                  "contribucion" => array:1 [
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                      "titulo" => "Sporotrichosis masquerading as pyoderma gangrenosum&#58; case report and review of 19 cases of sporotrichosis"
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                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "D&#46;R&#46; Byrd"
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                            2 => "L&#46;E&#46; Gibson"
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                      ]
                    ]
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                    0 => array:1 [
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              "identificador" => "bib0015"
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                            2 => "R&#46; de Vasconcellos Carvalhaes de Oliveira"
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                      "titulo" => "Estudio de 50 pacientes con esporotricosis&#46; Evaluaci&#243;n cl&#237;nica y de laboratorio"
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                        0 => array:2 [
                          "etal" => false
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                            2 => "P&#46; Lavalle"
                            3 => "J&#46; Barba-Rubio"
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                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "S&#46; Fonseca-Reyes"
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                            2 => "R&#46;C&#46; Miranda-Ackerman"
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                        0 => array:2 [
                          "etal" => false
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                            0 => "F&#46; Lauermann"
                            1 => "M&#46; Lyra"
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