Case Letter
Seborrheic-like dermatitis and liver dysfunction in an infant: signs of Langerhans cell histiocytosis
Dermatology Division, Department of Internal Medicine, Faculdade de Medicina de Ribeirão Preto, Universidade de São Paulo, Ribeirão Preto, SP, Brazil
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(B), Coalescent erythematous papules under hematic and meliceric crusts, extending from the temporal to the retroauricular region.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">This report describes the case of a one-year and five-months-old female child, who had desquamation and pruritus on the scalp for 6 months, with no response to topical corticosteroids and antifungal agents. The physical examination showed weight and height deficit (percentile 3 for height and 1 for weight); coalescent papules under hematic and meliceric crusts, on erythematous-desquamative skin on the scalp and the temporal and retroauricular regions (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>); ulcerated erythematous-infiltrated lesions on the left axilla and vulva and soft palate erosions. The liver was hardened, palpable five cm from the right costal margin. There was no adenomegaly. Complementary tests showed anemia (Hb: 9.8 mg/dL), liver function tests alterations (AST: 136 U/L, ALT: 152 U/L, ALP: 1821 U/L, GGT: 907.3 U/L); ultrasonography showed the liver at the upper limit of normality, heterogeneous echotexture and areas of periportal hyperechogenicity; magnetic resonance cholangiography and bone scintigraphy showed no alterations; histopathology of the skin showed a lichenoid infiltrate of Langerhans cells with diffuse immunohistochemical positivity for CD1a and S100 (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Thus, the diagnosis of Langerhans cell histiocytosis (LCH) with seborrheic-like dermatitis lesions and liver involvement was established. Oncological treatment was initiated with prednisone and vinblastine for 12 weeks, with no improvement of skin lesions. One month after the end of the induction phase, with continuous lesions on the scalp, a new histopathology analysis showed persistence of a lichenoid inflammatory infiltrate of Langerhans cells with diffuse positivity for CD1a, whereas the ultrasonography showed hepatic infiltration and splenomegaly. The continuation of chemotherapy with mercaptopurine, prednisone and vinblastine for 12 months was indicated (currently ongoing).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Historically, LCH has been subdivided into four syndromes (Letterer-Siwe, Hand-Schuller-Christian, eosinophilic granuloma, and Hashimoto-Pritzker); however, current evidence indicates that not all cases fit into these categories.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> Recently, LCH has been redefined as an inflammatory myeloid neoplasia, attributed to the activation of mutations in the mitogen-activated protein kinase (MAPK) pathway,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> with the <span class="elsevierStyleItalic">BRAF-V600E</span> gene mutation being the most prevalent one.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Clinically, it is categorized by the involvement of one or multiple systems, either unifocal or multifocal and the presence of involvement of organs at risk (liver, spleen and bone marrow), which define the prognosis and therapeutic response. Single-system LCH has an excellent prognosis, whereas the multi-system form has a mortality risk that requires aggressive treatments.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Although isolated skin disease is rare (2%), cutaneous manifestations are the standard form of presentation in around 80% of cases,<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2–4</span></a> being the most common in those younger than 2 years-old.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Seborrheic-like dermatitis, erythematous papules, and eczematous lesions are frequent,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> among others, including petechiae, purpura, maculae, hypopigmented or umbilicated papules, nodules, vesiculobullous lesions, and pustules,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> predominantly on the scalp, abdomen, chest and intertriginous areas. <a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The extracutaneous manifestations include lytic bone lesions, diabetes insipidus, growth hormone deficiency, hepatosplenomegaly, and lymphadenopathy.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Liver involvement is seen exclusively in multi-system LCH, presenting as isolated hepatomegaly and/or liver function impairment and jaundice.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The histopathological analysis and positivity for CD1a, S100, and/or CD207 (Langerin) in immunohistochemistry establishes the diagnosis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Cutaneous manifestations of LCH are variable and may be similar to other prevalent dermatoses. In the presence of intense and refractory seborrheic dermatitis-like condition, LCH should be suspected, and histopathological and multisystem involvement investigations are mandatory.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0025" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contributions</span><p id="par0030" class="elsevierStylePara elsevierViewall">Daniela Antoniali: Design and planning of the studied case; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied case; review of the literature; drafting and editing of the manuscript.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Helena Barbosa Lugão: Approval of the final version of the manuscript; drafting and editing of the manuscript; participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied case; critical review of the literature; critical review of the manuscript.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Daniel Elias: Approval of the final version of the manuscript; drafting and editing of the manuscript; participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied case; critical review of the literature; critical review of the manuscript.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Cacilda da Silva Souza: Approval of the final version of the manuscript; drafting and editing of the manuscript; collection, analysis, and interpretation of data; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied case; critical review of the literature; critical review of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors’ contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-07-05" "fechaAceptado" => "2020-08-11" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Dermatology Division, Department of Internal Medicine, Faculdade de Medicina de Ribeirão Preto, Universidade de São Paulo, Ribeirão Preto, SP, Brazil.</p>" ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1636 "Ancho" => 2507 "Tamanyo" => 374539 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A), Erythematous-desquamative, infiltrated lesions covered by hematic and meliceric crusts all over the scalp and the ear pinna. (B), Coalescent erythematous papules under hematic and meliceric crusts, extending from the temporal to the retroauricular region.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1210 "Ancho" => 3340 "Tamanyo" => 407605 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A), Spongiotic epidermis with foci of basal hydropic degeneration; superficial dermis with moderate diffuse band-like inflammatory infiltrate (Hematoxylin & eosin, ×100). (B), Cells with an oval nucleus, focally lobulated, in addition to mature lymphocytes and some eosinophils (Hematoxylin & eosin, ×200). (C), Diffuse positivity for CD1A (×200).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The multiple faces of Langerhans cell histiocytosis in childhood: A gentle reminder" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "M. Papadopoulou" 1 => "P. Panagopoulou" 2 => "A. Papadopoulou" 3 => "E. Hatzipantelis" 4 => "I. Efstratiou" 5 => "A. 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| Ano/Mês | Html | Total | |
|---|---|---|---|
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| 2024 Fevereiro | 141 | 82 | 223 |
| 2024 Janeiro | 114 | 64 | 178 |
| 2023 Dezembro | 102 | 49 | 151 |
| 2023 Novembro | 85 | 85 | 170 |
| 2023 Outubro | 59 | 78 | 137 |
| 2023 Setembro | 64 | 72 | 136 |
| 2023 Agosto | 48 | 37 | 85 |
| 2023 Julho | 59 | 40 | 99 |
| 2023 Junho | 64 | 28 | 92 |
| 2023 Maio | 55 | 25 | 80 |
| 2023 Abril | 47 | 21 | 68 |
| 2023 Março | 62 | 50 | 112 |
| 2023 Fevereiro | 41 | 35 | 76 |
| 2023 Janeiro | 57 | 74 | 131 |
| 2022 Dezembro | 63 | 44 | 107 |
| 2022 Novembro | 82 | 53 | 135 |
| 2022 Outubro | 113 | 63 | 176 |
| 2022 Setembro | 63 | 77 | 140 |
| 2022 Agosto | 45 | 47 | 92 |
| 2022 Julho | 41 | 57 | 98 |
| 2022 Junho | 60 | 62 | 122 |
| 2022 Maio | 43 | 52 | 95 |
| 2022 Abril | 60 | 58 | 118 |
| 2022 Março | 63 | 75 | 138 |
| 2022 Fevereiro | 104 | 51 | 155 |
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| 2021 Dezembro | 45 | 51 | 96 |
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