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and mild neutrophilia &#40;8980&#47;&#956;L&#41; without leukocytosis&#46; The chest radiograph revealed a cavitated nodule in the middle field of the left lung&#46; A review of the tests carried out in primary care revealed a fungal culture in which <span class="elsevierStyleItalic">Scedosporium apiospermum</span> complex was isolated&#44; with a minimum inhibitory concentration of 1 for voriconazole&#46; The patient was diagnosed with sporotrichoid lymphocutaneous fungal infection and admitted because of possible disseminated infection while immunosuppressed&#46; Treatment was started with oral voriconazole at 400<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h on day 1 and 200<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h on the following days&#46; Histopathology revealed a sinus tract filled with hyperkeratotic material&#44; with a pustule on the surface&#46; Periodic acid-Schiff staining revealed mytotic structures in the form of spores and hyphae with clear 45&#176; branching &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Tissue culture confirmed isolation of <span class="elsevierStyleItalic">S apiospermum</span> and was negative for bacteria and mycobacteria&#46; The antifungal susceptibility profile confirmed sensitivity to voriconazole&#46; The result of polymerase chain reaction assay with <span class="elsevierStyleItalic">Sporothrix schenckii</span> and mycobacteria was negative&#46; The patient&#39;s skin complaint progressed favorably&#44; with crusting lesions that replaced the pustules on the dorsum of the hand and ulcerated lesions instead of nodules on the forearm&#46; Computed tomography of the chest confirmed the presence of a cavitated nodule in the left lower lobe and other&#44; smaller nodules that were probably fungal in origin &#40;given the patient&#39;s baseline status and after agreement with his family&#44; we decided against further testing&#41;&#46; Nevertheless&#44; the possibility of endocarditis with septic embolism was ruled out using transthoracic echocardiography&#46; During admission&#44; the most remarkable observation was severe voriconazole-induced hyponatremia&#44; which resolved gradually with fluid and electrolyte therapy and temporary suspension of the drug&#46; Onset of self-limiting episodes of visual hallucinations necessitated cranial computed tomography&#44; which ruled out fungi in the parenchyma&#46; Therefore&#44; the episodes were considered part of a multifactorial confusional state&#46; The favorable laboratory outcome and clinical course led us to discharge the patient after reintroducing voriconazole&#46; The bullous and nodular lesions disappeared after 22 days of intravenous treatment&#44; leaving residual purpuric lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>C and D&#41;&#44; and the follow-up radiograph revealed complete cavitation of the lung nodule&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S apiospermum</span> is a ubiquitous mold found throughout the world&#46; It is isolated in rural soil&#44; contaminated water&#44; and cattle and bird excrement&#46; It mainly infects immunosuppressed patients&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> in whom it more frequently disseminates through the bloodstream&#46; The main routes of transmission are direct inoculation via the skin and inhalation of spores&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The main targets are the skin&#44; the lungs&#44; and the central nervous system&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The most widely reported skin manifestations are bullous necrotic purpura<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">3&#44;4</span></a> and the spirotrichoid form&#44;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">5&#44;6</span></a> both of which occurred together in the case we present&#46; A high index of suspicion&#44; early diagnosis&#44; and rapid initiation of treatment are essential if we are to improve a prognosis that is already poor&#46; Although histology was unable to identify the pathogen&#44; it was complemented with a positive culture &#40;whitish colonies that progress to grayish and brownish colonies &#91;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#93;&#41; in order to establish the diagnosis&#46; In addition&#44; the extension study reveals the presence of invasive fungal infection&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a> Therefore&#44; skin involvement is essential for identification of the condition and for the need to speed up therapeutic procedures &#40;see above&#41;&#46; Monotherapy with voriconazole is postulated as the antifungal drug of choice depending on the resistance detected&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> although dosing and duration of treatment have not been established&#46; Despite efforts made to date&#44; mortality continues to be high &#40;40&#37;-100&#37; depending on the series&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">8&#44;9</span></a></p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">We present a case of opportunistic infection by <span class="elsevierStyleItalic">S apiospermum</span>&#44; a little-known pathogen with a peculiar clinical presentation that has not yet been reported in Spain&#46; A multidisciplinary approach is essential owing to the poor baseline status of affected patients&#44; the need to use highly toxic drugs&#44; and the eventual associated systemic complications&#46; In this sense&#44; the dermatologist may play a fundamental role in early diagnosis and survival&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case and Research Letters
Bullous Necrotic Purpura Caused by Scedosporium apiospermum Presenting With a Sporotrichoid Pattern
Púrpura bullonecrótica de distribución esporotricoide por Scedosporium apiospermum
J. Company-Quirogaa,
Autor para correspondencia
j.companyquiroga@gmail.com

Corresponding author.
, C. Martínez-Morána, A. Morenob, J. Borbujoa
a Servicio de Dermatología, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, España
b Servicio de Anatomía Patológica, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, España
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and chronic lymphatic leukemia&#46; He was receiving treatment with insulin glargine and oxycodone&#47;naloxone&#46; He reported that his condition had worsened despite topical antifungal treatment and various oral antibiotics that were started by his primary care physician&#44; as well as local treatments&#46; He denied having fever and relevant systemic symptoms&#46; The examination revealed a well-defined erythematous-violaceous plaque on the dorsum of the left hand&#46; The plaque was slightly infiltrated&#44; with multiple pustular and crusted lesions on the surface&#44; and was spreading in a sporotrichoid pattern over the dorsum of the forearm&#44; where ulcerated lesions alternated with fluctuant violaceous nodules that were seeping purulent exudate &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> A and B&#41;&#46; The most notable findings of the laboratory workup performed in the emergency department were a minimum increase in C-reactive protein concentration &#40;0&#46;8<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; and mild neutrophilia &#40;8980&#47;&#956;L&#41; without leukocytosis&#46; The chest radiograph revealed a cavitated nodule in the middle field of the left lung&#46; A review of the tests carried out in primary care revealed a fungal culture in which <span class="elsevierStyleItalic">Scedosporium apiospermum</span> complex was isolated&#44; with a minimum inhibitory concentration of 1 for voriconazole&#46; The patient was diagnosed with sporotrichoid lymphocutaneous fungal infection and admitted because of possible disseminated infection while immunosuppressed&#46; Treatment was started with oral voriconazole at 400<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h on day 1 and 200<span class="elsevierStyleHsp" style=""></span>mg&#47;12<span class="elsevierStyleHsp" style=""></span>h on the following days&#46; Histopathology revealed a sinus tract filled with hyperkeratotic material&#44; with a pustule on the surface&#46; Periodic acid-Schiff staining revealed mytotic structures in the form of spores and hyphae with clear 45&#176; branching &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Tissue culture confirmed isolation of <span class="elsevierStyleItalic">S apiospermum</span> and was negative for bacteria and mycobacteria&#46; The antifungal susceptibility profile confirmed sensitivity to voriconazole&#46; The result of polymerase chain reaction assay with <span class="elsevierStyleItalic">Sporothrix schenckii</span> and mycobacteria was negative&#46; The patient&#39;s skin complaint progressed favorably&#44; with crusting lesions that replaced the pustules on the dorsum of the hand and ulcerated lesions instead of nodules on the forearm&#46; Computed tomography of the chest confirmed the presence of a cavitated nodule in the left lower lobe and other&#44; smaller nodules that were probably fungal in origin &#40;given the patient&#39;s baseline status and after agreement with his family&#44; we decided against further testing&#41;&#46; Nevertheless&#44; the possibility of endocarditis with septic embolism was ruled out using transthoracic echocardiography&#46; During admission&#44; the most remarkable observation was severe voriconazole-induced hyponatremia&#44; which resolved gradually with fluid and electrolyte therapy and temporary suspension of the drug&#46; Onset of self-limiting episodes of visual hallucinations necessitated cranial computed tomography&#44; which ruled out fungi in the parenchyma&#46; Therefore&#44; the episodes were considered part of a multifactorial confusional state&#46; The favorable laboratory outcome and clinical course led us to discharge the patient after reintroducing voriconazole&#46; The bullous and nodular lesions disappeared after 22 days of intravenous treatment&#44; leaving residual purpuric lesions &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figs&#46; 1</a>C and D&#41;&#44; and the follow-up radiograph revealed complete cavitation of the lung nodule&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S apiospermum</span> is a ubiquitous mold found throughout the world&#46; It is isolated in rural soil&#44; contaminated water&#44; and cattle and bird excrement&#46; It mainly infects immunosuppressed patients&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> in whom it more frequently disseminates through the bloodstream&#46; The main routes of transmission are direct inoculation via the skin and inhalation of spores&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The main targets are the skin&#44; the lungs&#44; and the central nervous system&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> The most widely reported skin manifestations are bullous necrotic purpura<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">3&#44;4</span></a> and the spirotrichoid form&#44;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">5&#44;6</span></a> both of which occurred together in the case we present&#46; A high index of suspicion&#44; early diagnosis&#44; and rapid initiation of treatment are essential if we are to improve a prognosis that is already poor&#46; Although histology was unable to identify the pathogen&#44; it was complemented with a positive culture &#40;whitish colonies that progress to grayish and brownish colonies &#91;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#93;&#41; in order to establish the diagnosis&#46; In addition&#44; the extension study reveals the presence of invasive fungal infection&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a> Therefore&#44; skin involvement is essential for identification of the condition and for the need to speed up therapeutic procedures &#40;see above&#41;&#46; Monotherapy with voriconazole is postulated as the antifungal drug of choice depending on the resistance detected&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> although dosing and duration of treatment have not been established&#46; Despite efforts made to date&#44; mortality continues to be high &#40;40&#37;-100&#37; depending on the series&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">8&#44;9</span></a></p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">We present a case of opportunistic infection by <span class="elsevierStyleItalic">S apiospermum</span>&#44; a little-known pathogen with a peculiar clinical presentation that has not yet been reported in Spain&#46; A multidisciplinary approach is essential owing to the poor baseline status of affected patients&#44; the need to use highly toxic drugs&#44; and the eventual associated systemic complications&#46; In this sense&#44; the dermatologist may play a fundamental role in early diagnosis and survival&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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