Case and Research Letters
Erythroderma as an Initial Presentation of Langerhans Cell Histiocytosis Involving the Sinus
Eritrodermia como forma de presentación inicial de histiocitosis sinusal de células de Langerhans
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Un mayor aumento (C [H-E ×<span class="elsevierStyleHsp" style=""></span>40]) permite ver en detalle los eosinófilos (flechas) y los núcleos elongados con hendiduras o plegamientos de las células de Langerhans (D [CD1a] ×<span class="elsevierStyleHsp" style=""></span>10). Expresión positiva de CD1a en las células de Langerhans.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "R. Ruiz-Villaverde, M. Galán-Gutierrez, F. Ramos-Pleguezuelos, J.M. Martinez de Victoria" "autores" => array:4 [ 0 => array:2 [ "nombre" => "R." "apellidos" => "Ruiz-Villaverde" ] 1 => array:2 [ "nombre" => "M." "apellidos" => "Galán-Gutierrez" ] 2 => array:2 [ "nombre" => "F." "apellidos" => "Ramos-Pleguezuelos" ] 3 => array:2 [ "nombre" => "J.M." "apellidos" => "Martinez de Victoria" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S1578219014001371" "doi" => "10.1016/j.adengl.2014.05.003" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219014001371?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731013003232?idApp=UINPBA000044" "url" => "/00017310/0000010500000006/v1_201406280127/S0001731013003232/v1_201406280127/es/main.assets" ] ] "itemAnterior" => array:19 [ "pii" => "S1578219014001383" "issn" => "15782190" "doi" => "10.1016/j.adengl.2014.05.004" "estado" => "S300" "fechaPublicacion" => "2014-07-01" "aid" => "921" "copyright" => "Elsevier España, S.L. and AEDV" "documento" => "simple-article" "crossmark" => 0 "subdocumento" => "cor" "cita" => "Actas Dermosifiliogr. 2014;105:628-30" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:2 [ "total" => 2151 "formatos" => array:3 [ "EPUB" => 41 "HTML" => 1658 "PDF" => 452 ] ] "en" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letters</span>" "titulo" => "Old World Leishmaniasis on the Ear Lobe: A Rare Site" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "628" "paginaFinal" => "630" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Infección del lóbulo auricular por <span class="elsevierStyleItalic">Leishmania</span> en el Viejo Mundo: una localización excepcional" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 562 "Ancho" => 1440 "Tamanyo" => 165423 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Front and side views. A, Erythematous infiltrated plaque of 16<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>25<span class="elsevierStyleHsp" style=""></span>mm on the left ear lobe with central ulceration and a seropurulent crust and coalescing pustules on the antihelix. B, Soft edema and hyperemic nodular infiltration (9.5<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleHsp" style=""></span>mm) of the right ear lobe.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "G. Blasco Morente, J. Rodríguez-Granger, J. Tercedor Sánchez, J.M. Latorre Fuentes" "autores" => array:4 [ 0 => array:2 [ "nombre" => "G." "apellidos" => "Blasco Morente" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Rodríguez-Granger" ] 2 => array:2 [ "nombre" => "J." "apellidos" => "Tercedor Sánchez" ] 3 => array:2 [ "nombre" => "J.M." 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Ruiz-Villaverde, M. Galán-Gutierrez, F. Ramos-Pleguezuelos, J.M. Martinez de Victoria" "autores" => array:4 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Ruiz-Villaverde" "email" => array:1 [ 0 => "ismenios@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "M." "apellidos" => "Galán-Gutierrez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "F." "apellidos" => "Ramos-Pleguezuelos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "J.M." "apellidos" => "Martinez de Victoria" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Complejo Hospitalario de Jaén, Jaén, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Complejo Hospitalario de Jaén, Jaén, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Eritrodermia como forma de presentación inicial de histiocitosis sinusal de células de Langerhans" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 772 "Ancho" => 1032 "Tamanyo" => 95755 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Uniform violaceous erythema affecting the entire trunk.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 56-year-old man with a history of chronic obstructive pulmonary disease and hypertension who was being treated with eprosartan/hydrochlorothiazide and atorvastatin/amlodipine. He presented with an eczematous, pruritic skin condition that had developed 2 months previously.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed coalescing erythematous plaques on the upper limbs, face, and the upper third of the trunk, affecting approximately 25% of the body surface. There was no palmoplantar or mucosal involvement (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A skin biopsy of the affected area showed a superficial perivascular dermatitis with eosinophils and intraepidermal necrotic keratinocytes, suggesting drug-induced skin disease. Blood tests revealed leukocytosis (15 330<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L), with a predominance of neutrophils (82.2%), and increases in the erythrocyte sedimentation rate and in the levels of C-reactive protein and immunoglobulin E (><span class="elsevierStyleHsp" style=""></span>5000 IU/mL).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">After diagnosis of drug-induced skin disease antihypertensive treatment was discontinued and a tapering dosage of prednisone was prescribed, starting at 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d. After 40 days of treatment the dose of corticosteroids was reduced to 10<span class="elsevierStyleHsp" style=""></span>mg/d due to the development of cushingoid habitus. Given the progression of the skin lesions to erythroderma, affecting almost 90% of the body surface, the patient was treated with acitretin (25<span class="elsevierStyleHsp" style=""></span>mg/d), which stabilized the symptoms and reduced the intensity of the erythroderma and pruritus. The patient's baseline hypertension was controlled by dietary measures after a relapse induced by temporary treatment with enalapril.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Laboratory workups and monthly imaging studies revealed no differences as compared with the initial findings. Eight months later eosinophilia was detected (20.8%; 2310<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L). A new skin biopsy revealed no additional findings and bone marrow aspiration was normal.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Weeks later the patient noticed an enlarged right inguinal lymph node of 2<span class="elsevierStyleHsp" style=""></span>cm in diameter. A computed tomography scan of the chest and abdomen revealed considerable enlargement of the mediastinal lymph nodes (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A) and bilateral axillary and inguinal lymph nodes of over 1<span class="elsevierStyleHsp" style=""></span>cm in diameter (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B). No hepatosplenomegaly, mesenteric lymphadenopathy, or bone alterations were observed. Histological examination of the inguinal node revealed that the paracortex contained cellular aggregates consisting of large cells with grooved nuclei containing compact chromatin, together with a large, eosinophilic cytoplasm. Microabscesses and occasional histiocytes and plasma cells were also observed. The cells were positive for S-100/CD1a and negative for myeloperoxidase and CD117, and had a low proliferative index. A histological diagnosis of Langerhans cell histiocytosis (LCH) involving the sinus was established (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a> A-D). Based on this diagnosis, acitretin treatment was discontinued and the patient began treatment with tapering dosages of vinblastine (intravenous bolus, starting at 6<span class="elsevierStyleHsp" style=""></span>mg/m<span class="elsevierStyleSup">2</span>) and oral prednisone (starting at 40<span class="elsevierStyleHsp" style=""></span>mg/m<span class="elsevierStyleSup">2</span>/d) for 6 and 4 weeks, respectively. This resulted in an improvement in the patient's baseline condition. The patient remains asymptomatic after 3 months of follow-up.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Erythroderma is defined as inflammation that affects at least 90% of the body surface. Its etiologic diagnosis is difficult.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The dermatologist must be alert to the possibility of an underlying neoplasm in cases of insidious onset, treatment resistance, or weight loss, and in cases with no prior history of skin disease.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Histology is nonspecific, and despite the presence of signs specific to the primary disease, etiologic diagnosis is often not achieved.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">LCH is a relatively uncommon entity characterized by oligoclonal proliferation of Langerhans cells. Its incidence is unknown<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and it is difficult to diagnose. Cutaneous presentations of LCH resembling pyoderma gangrenosum, annular granuloma, amebiasis, eruptive xanthomas, and flat warts, among other conditions, have been described.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> LCH can affect one or more organs; the number of affected organs has implications for prognosis and treatment. This condition has been reported in association with solid tumors, lymphomas, and leukemias, and a common origin of LCH and hematological malignancies has been widely debated.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Several therapeutic strategies are used in the management of LCH in adult patients, depending on the number of organs involved and the clinical course. In our case, even though only lymphatic involvement was initially observed, we opted for aggressive therapy, which significantly reduces morbidity and mortality according to the Evaluation and Treatment Guidelines of the Histiocyte Society.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Erythroderma as an initial presentation of LCH, as occurred in the present case, has been described in only one series to date.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The main difficulty in the present case was the length of time between the onset of erythroderma and lymphadenopathy; we established a definitive histological diagnosis of the latter through histological analyses, as all findings hitherto were nonspecific. We wish to stress the importance of repeated complementary testing and close follow-up with retesting in patients with erythrodermas with an atypical clinical course and poor therapeutic response.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ruiz-Villaverde R, Galán-Gutierrez M, Ramos-Pleguezuelos F, Martinez de Victoria J. Eritrodermia como forma de presentación inicial de histiocitosis sinusal de células de Langerhans. Actas Dermosifiliogr. 2014;105:630–632.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 772 "Ancho" => 1032 "Tamanyo" => 95755 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Uniform violaceous erythema affecting the entire trunk.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 598 "Ancho" => 1448 "Tamanyo" => 112586 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Enlargement of the mediastinal lymph nodes. B, Palpable left inguinal lymph node of 3<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1137 "Ancho" => 1532 "Tamanyo" => 746855 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A, Lymph nodes affected by Langerhans cell histiocytosis (hematoxylin-eosin, original magnification ×2). B, Subcapsular sinus occupied and expanded by a predominantly mononuclear cell population containing some eosinophils (hematoxylin-eosin, original magnification ×20). C and D, Higher magnification images showing the eosinophils (arrowheads in C; hematoxylin-eosin, original magnification ×40) and the grooves or folds in the elongated nuclei of the CD1a<span class="elsevierStyleSup">+</span> Langerhans cells (D; CD1a, original magnification ×10).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Eritrodermia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A. Boada García" 1 => "H. Domingo Herranz" 2 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 8 | 12 | 20 |
| 2024 October | 64 | 38 | 102 |
| 2024 September | 69 | 38 | 107 |
| 2024 August | 109 | 70 | 179 |
| 2024 July | 84 | 29 | 113 |
| 2024 June | 97 | 31 | 128 |
| 2024 May | 68 | 29 | 97 |
| 2024 April | 74 | 25 | 99 |
| 2024 March | 76 | 37 | 113 |
| 2024 February | 81 | 49 | 130 |
| 2024 January | 66 | 35 | 101 |
| 2023 December | 55 | 18 | 73 |
| 2023 November | 69 | 30 | 99 |
| 2023 October | 76 | 36 | 112 |
| 2023 September | 85 | 32 | 117 |
| 2023 August | 48 | 20 | 68 |
| 2023 July | 68 | 38 | 106 |
| 2023 June | 55 | 31 | 86 |
| 2023 May | 50 | 23 | 73 |
| 2023 April | 44 | 28 | 72 |
| 2023 March | 44 | 31 | 75 |
| 2023 February | 33 | 18 | 51 |
| 2023 January | 36 | 24 | 60 |
| 2022 December | 44 | 47 | 91 |
| 2022 November | 26 | 29 | 55 |
| 2022 October | 33 | 16 | 49 |
| 2022 September | 23 | 36 | 59 |
| 2022 August | 30 | 37 | 67 |
| 2022 July | 37 | 41 | 78 |
| 2022 June | 28 | 24 | 52 |
| 2022 May | 46 | 40 | 86 |
| 2022 April | 57 | 49 | 106 |
| 2022 March | 39 | 54 | 93 |
| 2022 February | 44 | 31 | 75 |
| 2022 January | 31 | 46 | 77 |
| 2021 December | 33 | 30 | 63 |
| 2021 November | 45 | 40 | 85 |
| 2021 October | 51 | 37 | 88 |
| 2021 September | 38 | 42 | 80 |
| 2021 August | 33 | 13 | 46 |
| 2021 July | 40 | 36 | 76 |
| 2021 June | 42 | 19 | 61 |
| 2021 May | 49 | 28 | 77 |
| 2021 April | 73 | 58 | 131 |
| 2021 March | 60 | 22 | 82 |
| 2021 February | 49 | 24 | 73 |
| 2021 January | 16 | 16 | 32 |
| 2020 December | 30 | 9 | 39 |
| 2020 November | 23 | 19 | 42 |
| 2020 October | 24 | 11 | 35 |
| 2020 September | 26 | 15 | 41 |
| 2020 August | 26 | 25 | 51 |
| 2020 July | 24 | 12 | 36 |
| 2020 June | 32 | 25 | 57 |
| 2020 May | 29 | 27 | 56 |
| 2020 April | 16 | 13 | 29 |
| 2020 March | 24 | 18 | 42 |
| 2020 February | 2 | 4 | 6 |
| 2020 January | 0 | 4 | 4 |
| 2019 December | 4 | 10 | 14 |
| 2019 November | 4 | 0 | 4 |
| 2019 October | 0 | 3 | 3 |
| 2019 September | 4 | 6 | 10 |
| 2019 August | 4 | 4 | 8 |
| 2019 July | 4 | 6 | 10 |
| 2019 June | 4 | 11 | 15 |
| 2019 May | 7 | 13 | 20 |
| 2019 April | 2 | 2 | 4 |
| 2019 March | 4 | 6 | 10 |
| 2019 February | 0 | 1 | 1 |
| 2019 January | 4 | 0 | 4 |
| 2018 December | 6 | 0 | 6 |
| 2018 October | 4 | 0 | 4 |
| 2018 September | 1 | 5 | 6 |
| 2018 August | 0 | 11 | 11 |
| 2018 June | 0 | 3 | 3 |
| 2018 May | 0 | 13 | 13 |
| 2018 April | 0 | 1 | 1 |
| 2018 March | 0 | 2 | 2 |
| 2018 February | 34 | 4 | 38 |
| 2018 January | 40 | 5 | 45 |
| 2017 December | 45 | 11 | 56 |
| 2017 November | 32 | 7 | 39 |
| 2017 October | 31 | 1 | 32 |
| 2017 September | 30 | 11 | 41 |
| 2017 August | 28 | 11 | 39 |
| 2017 July | 21 | 4 | 25 |
| 2017 June | 37 | 16 | 53 |
| 2017 May | 43 | 17 | 60 |
| 2017 April | 27 | 21 | 48 |
| 2017 March | 23 | 29 | 52 |
| 2017 February | 56 | 13 | 69 |
| 2017 January | 22 | 8 | 30 |
| 2016 December | 38 | 12 | 50 |
| 2016 November | 59 | 17 | 76 |
| 2016 October | 57 | 9 | 66 |
| 2016 September | 48 | 19 | 67 |
| 2016 August | 55 | 18 | 73 |
| 2016 July | 29 | 10 | 39 |
| 2016 June | 14 | 3 | 17 |
| 2016 May | 14 | 1 | 15 |
| 2016 April | 3 | 1 | 4 |
| 2016 March | 8 | 1 | 9 |
| 2016 February | 11 | 2 | 13 |
| 2016 January | 14 | 1 | 15 |
| 2015 December | 12 | 2 | 14 |
| 2015 November | 13 | 1 | 14 |
| 2015 October | 13 | 2 | 15 |
| 2015 September | 19 | 1 | 20 |
| 2015 August | 11 | 2 | 13 |
| 2015 July | 41 | 3 | 44 |
| 2015 June | 42 | 1 | 43 |
| 2015 May | 39 | 9 | 48 |
| 2015 April | 25 | 2 | 27 |
| 2015 March | 25 | 2 | 27 |
| 2015 February | 27 | 1 | 28 |
| 2015 January | 14 | 4 | 18 |
| 2014 December | 12 | 3 | 15 |
| 2014 November | 13 | 3 | 16 |
| 2014 October | 13 | 6 | 19 |
| 2014 August | 0 | 1 | 1 |
| 2014 July | 3 | 0 | 3 |
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