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also affecting the palmoplantar region and the oral cavity &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Therefore&#44; the diagnostic hypotheses of LCH&#44; congenital cytomegalovirus &#40;CMV&#41;&#44; leukemia cutis&#44; and severe combined immunodeficiency were suggested&#46; A biopsy was performed&#44; and serologies were requested &#40;HIV&#44; CMV&#44; rubella&#44; toxoplasmosis&#44; VDRL&#41; which were negative&#44; whereas screening for congenital immunodeficiency showed no alterations&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histopathology revealed chronic dermatitis associated with the presence of cells suggestive of Langerhans cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry showed positivity for CD1a&#44; CD68&#44; S100 protein&#59; Ki67 was positive in 70&#37; of the cells &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Once the diagnosis of LCH was confirmed&#44; the investigation of other organs through a myelogram and computed tomography of the chest&#44; abdomen&#44; and pelvis led to the classification of the case as multisystemic LCH due to pulmonary and hepatic involvement&#46; Treatment was started with weekly vinblastine 3&#8239;mg&#47;m<span class="elsevierStyleSup">2</span> &#43; prednisone 20&#8239;mg&#47;m<span class="elsevierStyleSup">2</span>&#44; according to the <span class="elsevierStyleItalic">Brazilian Society of Histiocytosis</span> guideline&#46; The condition improved significantly&#44; but after about two months the skin lesions returned&#44; and the pulmonary condition worsened&#44; requiring oxygen therapy&#46; A new chemotherapy regimen with Cladribine &#40;2-CdA&#41; was introduced&#44; according to the Japanese protocol&#46; The patient remains stable and is being followed by the oncology team&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The incidence of LCH ranges from two to nine cases per million children under 15 years of age&#44; 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with theories that support both a reactional and a neoplastic nature&#46; Characterization as a neoplastic disorder is supported by the finding of a mutation in the BRAF V600E gene&#44; and activation of the MAP kinase &#40;MAPK&#41; pathway&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The diagnosis depends on the clinical&#44; anatomopathological and immunohistochemical correlation&#46; Histopathologically&#44; varying amounts of Langerhans cells with a &#8220;coffee bean&#8221; appearance are observed&#46; Positive immunohistochemistry for CD1a and CD207 &#40;langerin&#41; establishes the diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Treatment will depend on disease extent and severity&#46; When the involvement is solely cutaneous&#44; spontaneous resolution is common&#46; For multisystem disease&#44; treatment with systemic steroids and vinblastine for 12 months is the first-line regimen&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">LCH is a serious disease that&#44; due to its very diverse clinical manifestation&#44; is often diagnosed late&#44; delaying treatment&#46; The presence of a dermatologist in the clinical staff allows its early identification&#44; with a significant impact on patient prognosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0065" class="elsevierStylePara elsevierViewall">Tha&#237;s Oliveira Utiyama&#58; 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Letter - Clinical
Langerhans cell histiocytosis: a rare case of the multisystemic form in an infant
Thaís Oliveira Utiyama
Autor para correspondência
thaisutiyama@hotmail.com

Corresponding author.
, Maria Laura Malzoni, Thalita Gabrieli Sanches Vasques, Cassiano Tamura Vieira Gomes
Faculty of Medical and Health Sciences of Sorocaba, Pontifícia Universidade Católica de São Paulo, Sorocaba, SP, Brazil
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Langerhans cell histiocytosis &#40;LCH&#41; is an inflammatory neoplasm of myeloid precursor cells&#44; in which there is an accumulation of specialized dendritic cells in different organs&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This case report describes a rare case of LCH with a multisystemic presentation&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">This case describes a two-month-old male patient&#44; presenting erythematous-purpuric lesions scattered throughout the body since birth&#46; After a short period of apparent improvement&#44; the lesions recurred&#46; There were no systemic manifestations&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">On physical examination&#44; angiomatous papules with hematic crusts on top and some flat&#44; hypochromic&#44; shiny papules were observed&#44; also affecting the palmoplantar region and the oral cavity &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Therefore&#44; the diagnostic hypotheses of LCH&#44; congenital cytomegalovirus &#40;CMV&#41;&#44; leukemia cutis&#44; and severe combined immunodeficiency were suggested&#46; A biopsy was performed&#44; and serologies were requested &#40;HIV&#44; CMV&#44; rubella&#44; toxoplasmosis&#44; VDRL&#41; which were negative&#44; whereas screening for congenital immunodeficiency showed no alterations&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Histopathology revealed chronic dermatitis associated with the presence of cells suggestive of Langerhans cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry showed positivity for CD1a&#44; CD68&#44; S100 protein&#59; Ki67 was positive in 70&#37; of the cells &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Once the diagnosis of LCH was confirmed&#44; the investigation of other organs through a myelogram and computed tomography of the chest&#44; abdomen&#44; and pelvis led to the classification of the case as multisystemic LCH due to pulmonary and hepatic involvement&#46; Treatment was started with weekly vinblastine 3&#8239;mg&#47;m<span class="elsevierStyleSup">2</span> &#43; prednisone 20&#8239;mg&#47;m<span class="elsevierStyleSup">2</span>&#44; according to the <span class="elsevierStyleItalic">Brazilian Society of Histiocytosis</span> guideline&#46; The condition improved significantly&#44; but after about two months the skin lesions returned&#44; and the pulmonary condition worsened&#44; requiring oxygen therapy&#46; A new chemotherapy regimen with Cladribine &#40;2-CdA&#41; was introduced&#44; according to the Japanese protocol&#46; The patient remains stable and is being followed by the oncology team&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The incidence of LCH ranges from two to nine cases per million children under 15 years of age&#44; with a peak between one and three years of age&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It can affect one or multiple organs&#44; with the following being considered at risk&#58; liver&#44; spleen&#44; and bone marrow&#46; Most patients have single-system involvement &#40;70&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The most frequently affected organs are the bones&#44; followed by the skin&#44; but in infants&#44; cutaneous manifestations are the main findings&#46; Dermatologically&#44; they present as a seborrheic-like dermatitis&#44; and less often as hemorrhagic lesions&#44; although these are favorable conducive to diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The pathogenesis remains unclear&#44; with theories that support both a reactional and a neoplastic nature&#46; Characterization as a neoplastic disorder is supported by the finding of a mutation in the BRAF V600E gene&#44; and activation of the MAP kinase &#40;MAPK&#41; pathway&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The diagnosis depends on the clinical&#44; anatomopathological and immunohistochemical correlation&#46; Histopathologically&#44; varying amounts of Langerhans cells with a &#8220;coffee bean&#8221; appearance are observed&#46; Positive immunohistochemistry for CD1a and CD207 &#40;langerin&#41; establishes the diagnosis&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Treatment will depend on disease extent and severity&#46; When the involvement is solely cutaneous&#44; spontaneous resolution is common&#46; For multisystem disease&#44; treatment with systemic steroids and vinblastine for 12 months is the first-line regimen&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">LCH is a serious disease that&#44; due to its very diverse clinical manifestation&#44; is often diagnosed late&#44; delaying treatment&#46; The presence of a dermatologist in the clinical staff allows its early identification&#44; with a significant impact on patient prognosis&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0065" class="elsevierStylePara elsevierViewall">Tha&#237;s Oliveira Utiyama&#58; Drafting and editing of the manuscript&#59; critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">Maria Laura Malzoni&#58; Effective participation in research orientation&#59; intellectual participation in the propaedeutic and&#47;or therapeutic conduct of the studied cases&#59; critical review of the manuscript&#59; approval of the final version of the manuscript&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Thalita Gabrieli Sanches Vasques&#58; Critical review of the manuscript&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Cassiano Tamura Vieira Gomes&#58; Approval of the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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