Case and Research Letters
Enfermedad de Rosai-Dorfman cutánea: una nueva presentación clínica
Cutaneous Rosai-Dorfman Disease: A Novel Clinical Presentation
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Conde, A.Y. Kim, R. de Miguel, C.H. Nousari" "autores" => array:4 [ 0 => array:4 [ "nombre" => "J.M." "apellidos" => "Conde" "email" => array:1 [ 0 => "docmoscoso@gmail.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" => "A.Y." "apellidos" => "Kim" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "R." "apellidos" => "de Miguel" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "C.H." "apellidos" => "Nousari" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Dermatology, Broward Health Medical Center/Nova Southeastern University College of Osteopathic Medicine, Fort Lauderdale, FL, EE.UU." "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Nova Southeastern University College of Osteopathic Medicine, Fort Lauderdale, FL, EE.UU." "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Celimar Central Clinic, West Bay, Islas Caimán" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Cutaneous Rosai-Dorfman Disease: A Novel Clinical Presentation" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 678 "Ancho" => 900 "Tamanyo" => 167588 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histopathological features. Dense nodular dermal mononuclear cell rich infiltrate showing a significant number of plasma cells and numerous scattered S100 positive multinucleated histiocytes with marked emperipolesis and inconspicuous eosinophils.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 21-year-old woman presented with a 3-month history of a diffuse asymptomatic eruption on her face and trunk. She denied fever or any other constitutional symptoms and her review of symptoms was non-contributory. She denied any pertinent past medical or family history. Her only medication included norgestimate/ethinyl estradiol which she had been taking for several years. Physical examination revealed diffuse scattered non-follicular based flesh-colored papules and small nodules. Many lesions demonstrated a central indentation resembling molluscum contagiosum (see <a class="elsevierStyleCrossRefs" href="#fig0005">Figures 1 & 2</a>). She did not have any cervical, axillary, or inguinal lymphadenopathy. Mucous membrane examination was unremarkable and lacrimal glands did not appear enlarged. Routine histologic examination of her right neck lesion revealed a dense nodular dermal mononuclear cell rich infiltrate showing a significant number of plasma cells and numerous scattered S100 positive multinucleated histiocytes with marked emperipolesis and inconspicuous eosinophils (see <a class="elsevierStyleCrossRef" href="#fig0015">Figure 3</a>). Complete blood count with differential, erythrocyte sedimentation rate, lactate dehydrogenase, liver transaminases, alkaline phosphatase, bilirubin, and creatinine were negative or within normal limits. Chest and abdominal magnetic resonance imaging were normal. She was treated with a 60<span class="elsevierStyleHsp" style=""></span>mg oral prednisone taper over six months which resulted in complete resolution of her skin lesions. Follow up at 10 months from onset of disease demonstrated complete remission.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Rosai-Dorfman Disease (RDD) is a relatively rare histiocytic proliferation disorder that was first described in 1969.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Though RDD classically presents with bilateral massive lymphadenopathy and systemic symptoms, it typically has a benign clinical course and favorable prognosis. In RDD, 43% of patients have involvement of other extranodal sites with skin being the most common site.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Though skin comprises 10% of extranodal sites involved, approximately 3% are solely cutaneous Rosai-Dorfman Disease (CRDD) without any nodal or other extranodal sites.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> While incidence is reportedly low, CRDD is more prevalent in middle-aged White and Asian women.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The etiology of RDD is unclear although immunologic,<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> viral, and genetic causes including SLC29A3 mutations have been hypothesized.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The classic presentation of CRDD is a relatively asymptomatic self-involuting nodulo-plaque with surrounding satellite papules.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> However, an evolving wide spectrum of clinical morphologic presentations have been reported. The most common site involved is the face, followed by thigh, and trunk. Recurrence has been reported to occur within 1 to 3.5 years.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> To the best of our knowledge, this will be the first description of RDD mimicking molluscum contagiosum.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The pathognomonic histologic finding of RDD is emperipolesis of intact lymphocytes by S100 and CD68 positive as well as CD1a and langerin negative pale histiocytes characterized also by vesicular nuclei and small nucleoli. Of note, emperipolesis entitles the presence of engulfing of intact hematologic cells, as opposed to hemophagiocytosis which degrades engulfed cells, by histiocytes or megakaryocytes. The so called “Rosai-Dorfman cell” is a type of histiocyte characterized by empiropolesis of only haloed lymphocytes.</p><p id="par0025" class="elsevierStylePara elsevierViewall">These characteristic histiocytes can be abundant or inconspicuously scattered amidst other inflammatory cells.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> Plasma cells are virtually present in all lesions of CRDD. Polymorphonuclear cells, namely eosinophils, are frequently found in skin lesions. Increased vascularity with plump endothelium, with or without fibrosis, are not uncommonly identified namely in long standing lesions.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Emperipolesis in cutaneous disorders has been traditionally related to RDD until recent descriptions of other disorders related to the so-called H syndrome.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> This disorder is characterized by hyperpigmentation, hypertrichosis, hearing loss, heart anomalies, hepatosplenomegaly, hypogonadism, and hallux valgus<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> and is caused by a mutation in the SLC29A3 gene. Many authors believe that syndromes associated with mutations in SLC29A3 including familial RDD and pigmented hypertrichosis with insulin-dependent diabetes,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> in addition to H syndrome, fall into the same spectrum of RDD.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Recognition of the pleomorphic genetic and phenotypic presentations of SCL29A3-related diseases is important for diagnosis and for consideration in the histopathologic differential diagnosis of emperipolesis. In addition to SCL29A3-related diseases, IgG4-related diseases have also been shown to have some overlap with RDD as a subset of RDD has been found to contain increased numbers of IgG4-positive plasma cells.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> However, much controversy remains regarding this.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Currently there is no standard guideline for the management of CRDD. The clinical course of CRDD is usually benign and self-limited. Spontaneous resolution varies and ranges from months to several years.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> A wide spectrum of therapeutic interventions including surgical excision, cryotherapy, radiotherapy, lesional and systemic corticosteroids, thalidomide, methotrexate, and even chemotherapy<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> have also been reported to be successful for resistant and/or recurrent lesions.</p><p id="par0045" class="elsevierStylePara elsevierViewall">In summary, we first present a case of CRDD with a peculiar eruption resembling molluscum contagiousum thus expanding the spectrum of the cutaneous clinical presentation of the disease and making CRDD another dermatologic masquerader. The nonspecific clinical presentation along with the not infrequently inconspicuous finding of Rosai-Dorfman cells in skin samples and furthermore the typical spontaneous resolution may represent clinico-pathologic characteristics that lead to the tangible possibility of CRDD being underreported.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:1 [ "titulo" => "Références" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Conde JM, Kim AY, de Miguel R, Nousari CH. Enfermedad de Rosai-Dorfman cutánea: una nueva presentación clínica. Actas Dermosifiliogr. 2018;109:655–657.</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" => 1200 "Ancho" => 900 "Tamanyo" => 110225 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Diffuse scattered non-follicular based flesh-colored papules and small nodules, some with central indentations, were seen on the face and the 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" => 675 "Ancho" => 900 "Tamanyo" => 66742 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Diffuse scattered non-follicular based flesh-colored papules and small nodules, some with central indentations, were seen on the face and the trunk.</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" => 678 "Ancho" => 900 "Tamanyo" => 167588 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histopathological features. Dense nodular dermal mononuclear cell rich infiltrate showing a significant number of plasma cells and numerous scattered S100 positive multinucleated histiocytes with marked emperipolesis and inconspicuous eosinophils.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "Références" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sinus histiocytosis with massive lymphadenopathy: A newly recognized benign clinicopathological entity" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J. Rosai" 1 => "R.F. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 9 | 16 |
| 2024 October | 71 | 44 | 115 |
| 2024 September | 68 | 29 | 97 |
| 2024 August | 83 | 52 | 135 |
| 2024 July | 66 | 31 | 97 |
| 2024 June | 78 | 23 | 101 |
| 2024 May | 84 | 34 | 118 |
| 2024 April | 88 | 27 | 115 |
| 2024 March | 73 | 22 | 95 |
| 2024 February | 67 | 32 | 99 |
| 2024 January | 53 | 25 | 78 |
| 2023 December | 61 | 15 | 76 |
| 2023 November | 90 | 34 | 124 |
| 2023 October | 71 | 38 | 109 |
| 2023 September | 71 | 40 | 111 |
| 2023 August | 60 | 18 | 78 |
| 2023 July | 67 | 50 | 117 |
| 2023 June | 55 | 30 | 85 |
| 2023 May | 63 | 24 | 87 |
| 2023 April | 43 | 23 | 66 |
| 2023 March | 85 | 26 | 111 |
| 2023 February | 57 | 34 | 91 |
| 2023 January | 51 | 44 | 95 |
| 2022 December | 50 | 59 | 109 |
| 2022 November | 51 | 21 | 72 |
| 2022 October | 34 | 13 | 47 |
| 2022 September | 39 | 44 | 83 |
| 2022 August | 26 | 43 | 69 |
| 2022 July | 31 | 33 | 64 |
| 2022 June | 22 | 28 | 50 |
| 2022 May | 43 | 40 | 83 |
| 2022 April | 50 | 42 | 92 |
| 2022 March | 49 | 51 | 100 |
| 2022 February | 39 | 36 | 75 |
| 2022 January | 49 | 49 | 98 |
| 2021 December | 38 | 42 | 80 |
| 2021 November | 34 | 49 | 83 |
| 2021 October | 44 | 61 | 105 |
| 2021 September | 38 | 45 | 83 |
| 2021 August | 33 | 40 | 73 |
| 2021 July | 26 | 40 | 66 |
| 2021 June | 26 | 34 | 60 |
| 2021 May | 44 | 62 | 106 |
| 2021 April | 132 | 95 | 227 |
| 2021 March | 74 | 54 | 128 |
| 2021 February | 84 | 59 | 143 |
| 2021 January | 50 | 32 | 82 |
| 2020 December | 50 | 33 | 83 |
| 2020 November | 42 | 43 | 85 |
| 2020 October | 25 | 15 | 40 |
| 2020 September | 44 | 31 | 75 |
| 2020 August | 22 | 28 | 50 |
| 2020 July | 24 | 19 | 43 |
| 2020 June | 32 | 44 | 76 |
| 2020 May | 31 | 29 | 60 |
| 2020 April | 13 | 16 | 29 |
| 2020 March | 17 | 16 | 33 |
| 2020 February | 0 | 1 | 1 |
| 2019 May | 1 | 0 | 1 |
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