Cases and Research Letters
Generalized Eruptive Histiocytosis in a Patient with Chronic Myelomonocytic Leukemia
Histiocitosis eruptiva generalizada en paciente con leucemia mielomonocítica crónica
I. Montero
, E. Gutiérrez-González, M. Ginarte, J. Toribio
Corresponding author
Departamento de Dermatología, Complejo Hospitalario Universitario, Facultad de Medicina, Santiago de Compostela, Spain
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Multiple spotty erythematous plaques on the lower lip, mulberry-like in appearance.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "B. Di MartinoOrtiz, M.L. Rodríguez-Oviedo, M. Rodríguez-Masi" "autores" => array:3 [ 0 => array:2 [ "nombre" => "B." "apellidos" => "Di MartinoOrtiz" ] 1 => array:2 [ "nombre" => "M.L." "apellidos" => "Rodríguez-Oviedo" ] 2 => array:2 [ "nombre" => "M." 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Montero, E. Gutiérrez-González, M. Ginarte, J. Toribio" "autores" => array:4 [ 0 => array:4 [ "nombre" => "I." "apellidos" => "Montero" "email" => array:1 [ 0 => "iriamontero@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "E." "apellidos" => "Gutiérrez-González" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Ginarte" ] 3 => array:2 [ "nombre" => "J." "apellidos" => "Toribio" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Departamento de Dermatología, Complejo Hospitalario Universitario, Facultad de Medicina, Santiago de Compostela, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Histiocitosis eruptiva generalizada en paciente con leucemia mielomonocítica crónica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 629 "Ancho" => 951 "Tamanyo" => 180988 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Infiltrate consisting of histiocytes, some of which are vacuolated, lymphocytes, and some multinucleated giant cells (hematoxylin-eosin, original magnification <span class="elsevierStyleHsp" style=""></span>×250).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">An 80-year-old man visited our department with asymptomatic skin lesions that had appeared 2 months earlier. The patient stated that he was not receiving any regular treatments and had not been administered new drugs; he reported no fever or other symptoms and did not recall having been bitten or stung.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed erythematous papules on the upper part of the torso; the papules measured approximately 3<span class="elsevierStyleHsp" style=""></span>mm in diameter and were firm with a depressed center (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). Some of the papules had coalesced to form plaques. The mucosa, palms, and soles were not affected.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A skin biopsy revealed a dense infiltrate in the papillary and middle dermis, consisting mainly of histiocytes, some of which were vacuolated, lymphocytes, and some multinucleated giant cells (<a class="elsevierStyleCrossRefs" href="#fig0015">Figs. 3 and 4</a>). Immune staining was positive for CD68 and negative for S100, CD1a, and factor XIIIa.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Laboratory tests and x-ray studies showed only monocytosis, which had already been diagnosed and was being followed up by the hematology department after a diagnosis of chronic myelomonocytic leukemia with limited bone marrow dysplasia and a normal karyotype.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was diagnosed with generalized eruptive histiocytosis and a wait-and-see approach was adopted; the lesions disappeared 6 months after diagnosis. The chronic myelomonocytic leukemia has remained stable to date.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Histiocytosis comprises a rare, heterogeneous group of diseases characterized by the proliferation of histiocytes. Most authors continue to use the traditional classification of the entity into Langerhans cell histiocytosis, type I histiocytosis or histiocytosis X, and non-Langerhans cell, type II, or non-X histiocytosis.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The group of non-Langerhans cell histiocytoses includes multiple diseases, such as generalized eruptive histiocytosis. Some authors maintain that these are not independent diseases but are different stages in the course of the same disease, and that generalized eruptive histiocytosis is the initial stage of the process, with subsequent development of xanthomatous lesions.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In 1963, Winkelmann and Muller<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> described generalized eruptive histiocytosis as a rare condition characterized by multiple noncoalescing, symmetric erythematous papules distributed on the torso and limbs. The disease is not accompanied by extracutaneous involvement and histology shows a monomorphic infiltrate consisting predominantly of histiocytes that are negative for S100 and lipids. The disease has a benign course and mainly affects middle-aged patients. It tends to remit spontaneously and, although the cause has not been established, some authors maintain that it is produced by the apoptosis mechanism.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">In our patient, the differential diagnosis included generalized granuloma anulare, which was ruled out, and immune staining indicated a diagnosis of a type of non-Langerhans cell histiocytosis. The clinical appearance of the lesions, their location, and the patient's age at onset ruled out other types non-Langerhans cell histiocytosis, such as benign cephalic histiocytosis, multicentric reticulohistiocytosis, and progressive nodular histiocytoma. The principal differential diagnosis includes other forms of xanthomatous non-Langerhans cell histiocytosis, particularly adult papular xanthoma. Adult papular xanthoma may be a later stage in the development of the disease, in which the clinical lesions take on a yellowish color and are characterized histologically by a greater number of xanthomatous cells.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">A wait-and-see approach is usually adopted, as the condition tends to resolve spontaneously within a few months. Treatment should be considered when remission does not occur and good results have been reported for systemic treatment with corticosteroids, hydroxychloroquine, and thalidomide,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> with psoralen-UVA<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> or even cryotherapy, peeling, or carbon dioxide laser treatment in localized lesions.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Our patient presented 3 unique aspects: coalescence of the lesions, negative immune staining for factor XIIIa, and association with chronic myelomonocytic leukemia.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Coalescence of the lesions has been reported in the late stage of this disease. Klemke et al.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> linked this clinical sign in this type of histiocytosis with acute monocytic leukemia and with a worse prognosis for the disease. Hence, the coalescence of the lesions in our patient and the myelomonocytic leukemia support this observation.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Another characteristic of our patient is the negative immune staining for factor XIIIa, which is usually positive in generalized eruptive histiocytosis; 2 other cases have been reported in the literature with this characteristic.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,8</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In general, non-Langerhans cell histiocytosis presents a benign course in healthy patients and does not usually appear in association with malignant diseases. However, some types of non-Langerhans cell histiocytosis, such as progressive nodular histiocytosis and juvenile xanthogranuloma, have been described in association with hematologic disorders.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> To date, 2 cases have been reported in the literature of generalized eruptive histiocytosis associated with acute monocytic leukemia,<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,10</span></a> but none have been described in association with chronic myelomonocytic leukemia, as was the case with our patient.</p><p id="par0075" class="elsevierStylePara elsevierViewall">In conclusion, we report what we believe to be the first case of a patient with generalized eruptive histiocytosis associated with chronic myelomonocytic leukemia. In our opinion, given the frequency of hematologic disorders in patients with non-Langerhans cell histiocytosis, further study of this association is needed in patients with these signs and symptoms, and more specifically, in patients with generalized eruptive histiocytosis with coalescing lesions.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Montero I, Gutiérrez-González R, Ginarte M, Toribio J. Histiocitosis eruptive generalizada en paciente con leukemia mielomonocítica crónica. Actas Dermosifiliogr. 2012;103:643–644.</p>" ] ] "multimedia" => array:4 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1273 "Ancho" => 951 "Tamanyo" => 167067 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple erythematous papules distributed on the patient's torso; some of the papules have coalesced to form small plaques.</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" => 711 "Ancho" => 951 "Tamanyo" => 124610 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Detail of the lesions, showing millimeter-sized umbilicated papules.</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" => 631 "Ancho" => 951 "Tamanyo" => 237561 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Dense infiltrate in the papillary and middle dermis (hematoxylin-eosin, original magnification <span class="elsevierStyleHsp" style=""></span>×100).</p>" ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 629 "Ancho" => 951 "Tamanyo" => 180988 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Infiltrate consisting of histiocytes, some of which are vacuolated, lymphocytes, and some multinucleated giant cells (hematoxylin-eosin, original magnification <span class="elsevierStyleHsp" style=""></span>×250).</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" => "Benign non-X histiocytosis: a unique case bridging several of the non-X histiocytic syndromes" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "B.M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 17 | 9 | 26 |
| 2024 October | 80 | 40 | 120 |
| 2024 September | 101 | 29 | 130 |
| 2024 August | 113 | 61 | 174 |
| 2024 July | 94 | 31 | 125 |
| 2024 June | 102 | 34 | 136 |
| 2024 May | 81 | 34 | 115 |
| 2024 April | 69 | 26 | 95 |
| 2024 March | 94 | 36 | 130 |
| 2024 February | 80 | 32 | 112 |
| 2024 January | 73 | 35 | 108 |
| 2023 December | 78 | 19 | 97 |
| 2023 November | 68 | 25 | 93 |
| 2023 October | 60 | 16 | 76 |
| 2023 September | 71 | 29 | 100 |
| 2023 August | 63 | 34 | 97 |
| 2023 July | 79 | 42 | 121 |
| 2023 June | 69 | 29 | 98 |
| 2023 May | 90 | 32 | 122 |
| 2023 April | 63 | 32 | 95 |
| 2023 March | 39 | 26 | 65 |
| 2023 February | 50 | 24 | 74 |
| 2023 January | 58 | 42 | 100 |
| 2022 December | 48 | 36 | 84 |
| 2022 November | 25 | 19 | 44 |
| 2022 October | 38 | 19 | 57 |
| 2022 September | 25 | 24 | 49 |
| 2022 August | 33 | 36 | 69 |
| 2022 July | 29 | 28 | 57 |
| 2022 June | 30 | 24 | 54 |
| 2022 May | 76 | 37 | 113 |
| 2022 April | 100 | 52 | 152 |
| 2022 March | 80 | 64 | 144 |
| 2022 February | 49 | 29 | 78 |
| 2022 January | 50 | 39 | 89 |
| 2021 December | 56 | 29 | 85 |
| 2021 November | 52 | 53 | 105 |
| 2021 October | 47 | 51 | 98 |
| 2021 September | 45 | 37 | 82 |
| 2021 August | 53 | 24 | 77 |
| 2021 July | 30 | 15 | 45 |
| 2021 June | 50 | 34 | 84 |
| 2021 May | 41 | 25 | 66 |
| 2021 April | 65 | 63 | 128 |
| 2021 March | 69 | 23 | 92 |
| 2021 February | 68 | 22 | 90 |
| 2021 January | 36 | 25 | 61 |
| 2020 December | 45 | 12 | 57 |
| 2020 November | 29 | 8 | 37 |
| 2020 October | 26 | 4 | 30 |
| 2020 September | 21 | 8 | 29 |
| 2020 August | 18 | 16 | 34 |
| 2020 July | 35 | 12 | 47 |
| 2020 June | 37 | 24 | 61 |
| 2020 May | 36 | 22 | 58 |
| 2020 April | 29 | 17 | 46 |
| 2020 March | 40 | 25 | 65 |
| 2020 February | 5 | 2 | 7 |
| 2020 January | 3 | 3 | 6 |
| 2019 December | 6 | 2 | 8 |
| 2019 November | 4 | 5 | 9 |
| 2019 October | 0 | 8 | 8 |
| 2019 September | 0 | 2 | 2 |
| 2019 August | 6 | 4 | 10 |
| 2019 July | 4 | 3 | 7 |
| 2019 June | 4 | 1 | 5 |
| 2019 May | 4 | 16 | 20 |
| 2019 April | 2 | 3 | 5 |
| 2019 March | 2 | 6 | 8 |
| 2019 January | 3 | 1 | 4 |
| 2018 December | 2 | 0 | 2 |
| 2018 November | 1 | 1 | 2 |
| 2018 October | 4 | 0 | 4 |
| 2018 September | 4 | 0 | 4 |
| 2018 June | 0 | 1 | 1 |
| 2018 May | 0 | 6 | 6 |
| 2018 March | 8 | 4 | 12 |
| 2018 February | 42 | 4 | 46 |
| 2018 January | 58 | 6 | 64 |
| 2017 December | 55 | 8 | 63 |
| 2017 November | 49 | 7 | 56 |
| 2017 October | 39 | 4 | 43 |
| 2017 September | 40 | 11 | 51 |
| 2017 August | 63 | 8 | 71 |
| 2017 July | 60 | 5 | 65 |
| 2017 June | 66 | 22 | 88 |
| 2017 May | 68 | 15 | 83 |
| 2017 April | 72 | 10 | 82 |
| 2017 March | 56 | 21 | 77 |
| 2017 February | 41 | 19 | 60 |
| 2017 January | 51 | 12 | 63 |
| 2016 December | 57 | 16 | 73 |
| 2016 November | 70 | 12 | 82 |
| 2016 October | 108 | 20 | 128 |
| 2016 September | 130 | 10 | 140 |
| 2016 August | 54 | 9 | 63 |
| 2016 July | 34 | 12 | 46 |
| 2016 June | 8 | 11 | 19 |
| 2016 May | 4 | 13 | 17 |
| 2016 April | 7 | 1 | 8 |
| 2016 March | 13 | 1 | 14 |
| 2016 February | 5 | 5 | 10 |
| 2016 January | 8 | 1 | 9 |
| 2015 December | 6 | 16 | 22 |
| 2015 November | 20 | 3 | 23 |
| 2015 October | 18 | 22 | 40 |
| 2015 September | 4 | 7 | 11 |
| 2015 August | 17 | 1 | 18 |
| 2015 July | 81 | 11 | 92 |
| 2015 June | 61 | 7 | 68 |
| 2015 May | 70 | 16 | 86 |
| 2015 April | 74 | 18 | 92 |
| 2015 March | 72 | 5 | 77 |
| 2015 February | 63 | 7 | 70 |
| 2015 January | 70 | 13 | 83 |
| 2014 December | 52 | 8 | 60 |
| 2014 November | 54 | 11 | 65 |
| 2014 October | 113 | 14 | 127 |
| 2014 September | 76 | 14 | 90 |
| 2014 August | 40 | 5 | 45 |
| 2014 July | 40 | 5 | 45 |
| 2014 June | 44 | 4 | 48 |
| 2014 May | 49 | 5 | 54 |
| 2014 April | 45 | 5 | 50 |
| 2014 March | 53 | 7 | 60 |
| 2014 February | 43 | 5 | 48 |
| 2014 January | 53 | 11 | 64 |
| 2013 December | 49 | 7 | 56 |
| 2013 November | 46 | 10 | 56 |
| 2013 October | 36 | 7 | 43 |
| 2013 September | 25 | 3 | 28 |
| 2013 August | 15 | 11 | 26 |
| 2013 July | 12 | 5 | 17 |
| 2013 June | 15 | 8 | 23 |
| 2013 May | 16 | 10 | 26 |
| 2013 April | 14 | 7 | 21 |
| 2013 March | 15 | 7 | 22 |
| 2013 February | 38 | 4 | 42 |
| 2013 January | 58 | 5 | 63 |
| 2012 December | 29 | 6 | 35 |
| 2012 November | 0 | 1 | 1 |
| 2012 October | 0 | 1 | 1 |
| 2012 September | 1 | 1 | 2 |
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