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or lymph node enlargement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">He had been previously submitted to an anatomopathological examination at another service whose report showed epidermis with acanthosis&#44; hyper and parakeratosis&#44; dense lymphohistioplasmocytic inflammatory infiltrate in the dermis&#44; outlining granulomas and numerous neutrophils &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Upon the patient&#8217;s admission&#44; the PCR of a lesion sample was requested&#44; with a positive result for <span class="elsevierStyleItalic">Leishmania braziliensis</span> and a positive Montenegro test &#40;6&#8239;mm&#41;&#46; The other serologies and complementary tests showed no abnormalities&#44; except for subclinical hypothyroidism and dyslipidemia&#46; The patient was admitted for treatment with amphotericin B deoxycholate &#40;total dose of 1&#46;8&#8239;g&#41; due to the extension and exuberance of the skin lesions&#44; with partial improvement of the lesions during follow-up &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; While hospitalized&#44; the patient had an increase in nitrogenous slag &#40;urea and creatinine&#41; and a bloodstream infection&#46; <span class="elsevierStyleItalic">S&#46; aureus</span> was isolated in blood cultures and he was treated with oxacycline&#46; After three months&#44; he underwent a new course of treatment&#44; this time with liposomal amphotericin B &#40;3g total dose&#41;&#44; showing significant improvement of the dermatological condition&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Two months after the second treatment&#44; the patient developed partial recurrence and in an attempt to prevent another prolonged hospitalization with multiple morbidities&#44; treatment with miltefosine 50&#8239;mg every 8&#8239;hours&#44; for 38 days&#44; was chosen&#44; with good response and tolerability &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; The patient maintained a good response at follow-up three months later with only residulal scars &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">American cutaneous leishmaniasis &#40;ACL&#41; is a public health problem that affects 85 countries&#44; with an annual record of approximately one million new cases per year worldwide&#46; In Brazil&#44; there are records of cases in all states&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Transmission to humans occurs through the bite of female sandflies&#44; especially of the genus <span class="elsevierStyleItalic">Lutzomyia</span>&#46; ACL is a spectral disease&#44; whose clinical presentation varies according to the parasite species and the host&#39;s immune response&#46; The main clinical forms that have been described are the cutaneous&#44; mucosal&#44; disseminated&#44; or diffuse anergic cutaneous form&#44; with highly variable clinical presentations&#44; which can generate disfiguring lesions and definitive deformities&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Disseminated leishmaniasis is characterized by lesion polymorphism&#44; which can include acneiform papules&#44; nodules&#44; or ulcers&#44; with at least 10 lesions in at least two non-contiguous body segments&#44; with mucosal involvement in most cases&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These disseminated lesions usually appear two to six weeks after the initial ulcerated lesion&#44; and the association with fever&#44; chills&#44; asthenia&#44; and nausea is frequent&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The main differential diagnoses are diffuse anergic cutaneous leishmaniasis&#44; in which the lesions are more infiltrated and show no ulcerations or mucosal involvement&#44; and cutaneous leishmaniasis with multiple lesions in immunosuppressed patients&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient did not develop systemic manifestations and did not have the classical lesions of disseminated or diffuse anergic leishmaniasis either&#44; thus he was classified as atypical cutaneous leishmaniasis&#44; with multiple lesions and a verrucous presentation&#46; Despite the exuberance of the lesions and the patient&#8217;s origin from an endemic area&#44; it took more than one year for the patient to undergo adequate investigation and treatment&#44; which highlights the need to alert the population and health professionals to the importance of an early diagnosis&#46; Atypical forms of ACL have become increasingly frequent&#44; particularly in endemic areas&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Moreover&#44; patients with Down syndrome can have innate and adaptive immune response abnormalities&#44; which could explain the rare presentation of the case reported herein&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The pentavalent antimony &#40;meglumine antimoniate&#41; is the first choice for treatment of ACL in Brazil&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a> However&#44; in patients with atypical&#44; severe cases or in those with more than 20 lesions&#44; amphotericin B is indicated&#44; due to the inadequate response to antimonials in these cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> The deoxycholate form is more accessible because it has a lower cost&#44; but it has more adverse effects &#8211; renal function impairment&#44; phlebitis&#44; electrolyte disturbances&#44; gastrointestinal symptoms&#46; Liposomal amphotericin B is usually better tolerated&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">ACL is considered one of the priority neglected diseases&#44; and despite the great advances in medicine in recent years&#44; the treatment of this disease remains a challenge for dermatologists&#44; due to the high toxicity of the available drugs and low cure rates&#44; often requiring multiple treatment cycles to achieve a satisfactory outcome&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Miltefosine may be a promising therapeutic option&#44; as it is administered orally and shows good efficacy and safety&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">It is extremely important to carry out more studies research on new drugs to treat this disease and many other neglected diseases&#44; aiming to reduce deformities&#44; complications&#44; and the impact on the quality of life of the affected patients&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Financial support</span><p id="par0045" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Authors&#39; contributions</span><p id="par0050" class="elsevierStylePara elsevierViewall">Jana Regis Novaes&#58; Drafting and editing of the manuscript&#59; intellectual participation in the conduct of the case&#59; review of the literature&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Luise Ribeiro Daltro&#58; Drafting and editing of the manuscript&#59; intellectual participation in the conduct of the case&#59; review of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Thiago Amparo Ferreira&#58; Intellectual participation in the conduct of the case&#59; review of the manuscript&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Paulo Roberto Lima Machado&#58; Intellectual participation in the conduct of the case&#59; review of the literature and review of the manuscript&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflicts of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Tropical/Infectoparasitary Dermatology
Exuberant case of verrucous cutaneous leishmaniasis
Jana Regis Novaes
Corresponding author
dra.jananovaes@gmail.com

Corresponding author.
, Luise Ribeiro Daltro, Thiago Amparo Ferreira, Paulo Roberto Lima Machado
Hospital Universitário Professor Edgard Santos, Universidade Federal da Bahia, Salvador, BA, Brazil
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    "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 28-year-old male with Down syndrome reported the onset of lesions on his back one year and three months before&#44; with progressive increase &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; There was no pruritus&#44; pain&#44; or associated systemic symptoms&#46; He reported previous treatment with itraconazole and terbinafine&#44; with no response&#46; The physical examination disclosed the presence of coalescent papules forming an extensive verrucous plaque on the back&#44; with prominent hyperkeratotic areas&#44; in addition to crusted and eroded lesions with an erythematous base&#46; He also had similar smaller lesions on his arms and left thigh&#46; There were no lesions in the nostrils&#44; oral cavity&#44; or lymph node enlargement&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">He had been previously submitted to an anatomopathological examination at another service whose report showed epidermis with acanthosis&#44; hyper and parakeratosis&#44; dense lymphohistioplasmocytic inflammatory infiltrate in the dermis&#44; outlining granulomas and numerous neutrophils &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Upon the patient&#8217;s admission&#44; the PCR of a lesion sample was requested&#44; with a positive result for <span class="elsevierStyleItalic">Leishmania braziliensis</span> and a positive Montenegro test &#40;6&#8239;mm&#41;&#46; The other serologies and complementary tests showed no abnormalities&#44; except for subclinical hypothyroidism and dyslipidemia&#46; The patient was admitted for treatment with amphotericin B deoxycholate &#40;total dose of 1&#46;8&#8239;g&#41; due to the extension and exuberance of the skin lesions&#44; with partial improvement of the lesions during follow-up &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; While hospitalized&#44; the patient had an increase in nitrogenous slag &#40;urea and creatinine&#41; and a bloodstream infection&#46; <span class="elsevierStyleItalic">S&#46; aureus</span> was isolated in blood cultures and he was treated with oxacycline&#46; After three months&#44; he underwent a new course of treatment&#44; this time with liposomal amphotericin B &#40;3g total dose&#41;&#44; showing significant improvement of the dermatological condition&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Two months after the second treatment&#44; the patient developed partial recurrence and in an attempt to prevent another prolonged hospitalization with multiple morbidities&#44; treatment with miltefosine 50&#8239;mg every 8&#8239;hours&#44; for 38 days&#44; was chosen&#44; with good response and tolerability &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; The patient maintained a good response at follow-up three months later with only residulal scars &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">American cutaneous leishmaniasis &#40;ACL&#41; is a public health problem that affects 85 countries&#44; with an annual record of approximately one million new cases per year worldwide&#46; In Brazil&#44; there are records of cases in all states&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Transmission to humans occurs through the bite of female sandflies&#44; especially of the genus <span class="elsevierStyleItalic">Lutzomyia</span>&#46; ACL is a spectral disease&#44; whose clinical presentation varies according to the parasite species and the host&#39;s immune response&#46; The main clinical forms that have been described are the cutaneous&#44; mucosal&#44; disseminated&#44; or diffuse anergic cutaneous form&#44; with highly variable clinical presentations&#44; which can generate disfiguring lesions and definitive deformities&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Disseminated leishmaniasis is characterized by lesion polymorphism&#44; which can include acneiform papules&#44; nodules&#44; or ulcers&#44; with at least 10 lesions in at least two non-contiguous body segments&#44; with mucosal involvement in most cases&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> These disseminated lesions usually appear two to six weeks after the initial ulcerated lesion&#44; and the association with fever&#44; chills&#44; asthenia&#44; and nausea is frequent&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The main differential diagnoses are diffuse anergic cutaneous leishmaniasis&#44; in which the lesions are more infiltrated and show no ulcerations or mucosal involvement&#44; and cutaneous leishmaniasis with multiple lesions in immunosuppressed patients&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient did not develop systemic manifestations and did not have the classical lesions of disseminated or diffuse anergic leishmaniasis either&#44; thus he was classified as atypical cutaneous leishmaniasis&#44; with multiple lesions and a verrucous presentation&#46; Despite the exuberance of the lesions and the patient&#8217;s origin from an endemic area&#44; it took more than one year for the patient to undergo adequate investigation and treatment&#44; which highlights the need to alert the population and health professionals to the importance of an early diagnosis&#46; Atypical forms of ACL have become increasingly frequent&#44; particularly in endemic areas&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Moreover&#44; patients with Down syndrome can have innate and adaptive immune response abnormalities&#44; which could explain the rare presentation of the case reported herein&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The pentavalent antimony &#40;meglumine antimoniate&#41; is the first choice for treatment of ACL in Brazil&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;5</span></a> However&#44; in patients with atypical&#44; severe cases or in those with more than 20 lesions&#44; amphotericin B is indicated&#44; due to the inadequate response to antimonials in these cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> The deoxycholate form is more accessible because it has a lower cost&#44; but it has more adverse effects &#8211; renal function impairment&#44; phlebitis&#44; electrolyte disturbances&#44; gastrointestinal symptoms&#46; Liposomal amphotericin B is usually better tolerated&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">ACL is considered one of the priority neglected diseases&#44; and despite the great advances in medicine in recent years&#44; the treatment of this disease remains a challenge for dermatologists&#44; due to the high toxicity of the available drugs and low cure rates&#44; often requiring multiple treatment cycles to achieve a satisfactory outcome&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Miltefosine may be a promising therapeutic option&#44; as it is administered orally and shows good efficacy and safety&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">It is extremely important to carry out more studies research on new drugs to treat this disease and many other neglected diseases&#44; aiming to reduce deformities&#44; complications&#44; and the impact on the quality of life of the affected patients&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Financial support</span><p id="par0045" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Authors&#39; contributions</span><p id="par0050" class="elsevierStylePara elsevierViewall">Jana Regis Novaes&#58; Drafting and editing of the manuscript&#59; intellectual participation in the conduct of the case&#59; review of the literature&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Luise Ribeiro Daltro&#58; Drafting and editing of the manuscript&#59; intellectual participation in the conduct of the case&#59; review of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Thiago Amparo Ferreira&#58; Intellectual participation in the conduct of the case&#59; review of the manuscript&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Paulo Roberto Lima Machado&#58; Intellectual participation in the conduct of the case&#59; review of the literature and review of the manuscript&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflicts of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Article information
ISSN: 03650596
Original language: English
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