Case and research letter
Primary Cutaneous CD4+ Small- to Medium-Sized Pleomorphic T-Cell Lymphoma: Report of a Case With Spontaneous Resolution
Linfoma cutáneo primario de células T pleomórficas de pequeño y mediano tamaño CD4+: a propósito de un caso con resolución espontánea
a Servicio de Dermatología, Instituto Valenciano de Oncología, Valencia, Spain
b Servicio de Dermatología, Hospital Arnau de Vilanova, Valencia, Spain
c Servicio de Dermatología del Hospital General Universitario de Valencia
d Servicio de Anatomía Patológica, Hospital Arnau de Vilanova, Valencia, Spain
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Tumoración cutánea localizada en la mejilla izquierda, marrón-eritematosa, de superficie lisa y brillante. B. Completa resolución de la lesión 6 semanas después. Persiste hiperpigmentación residual y una zona central deprimida que se corresponde con el punto de la biopsia.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "F. Messeguer, E. Gimeno, A. Agusti-Mejias, J. San Juan" "autores" => array:4 [ 0 => array:2 [ "nombre" => "F." "apellidos" => "Messeguer" ] 1 => array:2 [ "nombre" => "E." "apellidos" => "Gimeno" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Agusti-Mejias" ] 3 => array:2 [ "nombre" => "J. 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Messeguer, E. Gimeno, A. Agusti-Mejias, J. San Juan" "autores" => array:4 [ 0 => array:4 [ "nombre" => "F." "apellidos" => "Messeguer" "email" => array:1 [ 0 => "francescmb@comv.es" ] "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" => "E." "apellidos" => "Gimeno" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Agusti-Mejias" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "J. San" "apellidos" => "Juan" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Instituto Valenciano de Oncología, Valencia, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Arnau de Vilanova, Valencia, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Dermatología del Hospital General Universitario de Valencia" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Arnau de Vilanova, Valencia, Spain" "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfoma cutáneo primario de células T pleomórficas de pequeño y mediano tamaño CD4+: a propósito de un caso con resolución espontánea" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 714 "Ancho" => 952 "Tamanyo" => 273488 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Dense dermal infiltrate consisting of small- and medium-sized pleomorphic lymphocytes and histiocytes (hematoxylin–eosin, original magnification x250).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Primary cutaneous CD4+ small- to medium-sized pleomorphic T-cell lymphoma is included as a provisional entity in the group of primary cutaneous peripheral T-cell lymphomas, both in the latest WHO classification of tumors of hematopoietic and lymphoid tissues and in the WHO-EORTC classification for cutaneous lymphomas.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> This lymphoma is defined by a predominance of CD4+ small- to medium-sized pleomorphic T cells, with no prior history of macules or plaques that would suggest mycosis fungoides.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 35-year-old man visited our department with an asymptomatic lesion on his left cheek; the lesion had appeared 4 weeks earlier and had grown rapidly. The only relevant information in the patient's past history was that he had taken allopurinol for 3 months to treat asymptomatic hyperuricemia. He had interrupted this medication 3 days before visiting our department. Physical examination revealed an oval, dome-shaped erythematous tumor measuring 3 x 4<span class="elsevierStyleHsp" style=""></span>cm on the left cheek. The tumor had well-defined borders and a shiny surface and was indurated on palpation (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">A biopsy of the lesion was taken for histopathology study. The low-magnification view showed that the epidermis was not involved and revealed a dense cellular infiltrate occupying the entire thickness of the dermis and reaching the subcutaneous tissue. The neoplastic infiltrate consisted predominantly of small- to medium-sized lymphocytes with moderate pleomorphism and mild atypia. Numerous histiocytes were also found among the lymphocytes (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Immunohistochemistry showed the cells to be strongly positive for CD3, CD4, and CD5. Furthermore, staining was focally positive for CD8, CD20, and CD68, but negative for CD7 and CD30. The analysis of T-cell clonality using PCR for the T-cell receptor gamma (TCRG) gene showed monoclonal rearrangement. On the basis of these data we established a diagnosis of primary cutaneous CD4+ small- to medium-sized pleomorphic T-cell lymphoma.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Further tests included an analysis of lymphocyte populations and immunoglobulin levels, computed tomography of the neck, thorax, abdomen, and pelvis, and bone-marrow biopsy. All results were normal or negative. The lesion underwent a rapid spontaneous reduction in size and was completely flat after 6 weeks of follow-up (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). The area of residual hyperpigmentation was not infiltrated or hard to the touch. The patient subsequently underwent local radiation therapy using a 6-MeV electron beam, with a total dose of 4000 cGy administered at a dosage of 200 cGy per day. He remained asymptomatic at 7 months (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Primary cutaneous CD4+ small- to medium-sized pleomorphic T-cell lymphoma is a rare disease and accounts for just 2% of all primary cutaneous lymphomas.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Clinical presentation is usually as a solitary plaque or tumor, typically located on the face, neck, or upper torso; it is asymptomatic and fast-growing.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–6</span></a> Spontaneous total remission of the lesions, as occurred with our patient, has been previously reported in only 1 case.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Histology reveals dense nodular or diffuse dermal infiltrates that tend to infiltrate the subcutaneous tissue. Focal epidermotropism is a possible finding and means that a diagnosis of mycosis fungoides should always be considered.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,6,7</span></a> By definition, there is a predominance of CD4+ small- to medium-sized pleomorphic lymphocytes, although up to 30% of large pleomorphic lymphocytes may be found. Cases where a higher percentage of large cells is found should be classified as peripheral T-cell lymphoma, not otherwise specified. Mixed inflammatory infiltrates have also been reported, consisting of histiocytes, B cells, plasma cells, and, in some cases, CD8+ T cells and eosinophils.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8,9</span></a> TCRG analysis shows monoclonal rearrangement in 60% to 70% of cases.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The differential diagnosis should principally include benign lymphoid proliferations (T-cell pseudolymphoma, pseudolymphomatous folliculitis, papules, nodules, and solitary lymphoid tumors) and mycosis fungoides in its unilesional and granulomatous forms.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> In our case, the rapid, self-limiting course, together with the fact that the patient had previously been treated with allopurinol, led to an initial diagnosis of T-cell pseudolymphoma. The subsequent pathology report made it possible to establish the diagnosis of primary cutaneous CD4+ small- to medium-sized pleomorphic T-cell lymphoma.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The prognosis in these lymphomas is generally very favorable, particularly in cases of solitary lesions, and 5-year survival is around 80%.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3,5</span></a> These cases of localized disease are usually treated with surgical excision or local radiation therapy, and more aggressive treatments such as chemotherapy are not required.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3,6,8</span></a> However, careful monitoring with regular follow-up visits is recommended.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The usually painless course and the clinical and histologic similarity that this type of lymphoma may bear to other cutaneous lymphoid infiltrates has led some authors to suggest the term <span class="elsevierStyleItalic">lymphocyte proliferations of uncertain significance</span> to refer to cases, without forcing a diagnosis, where it is not possible to determine whether the process is benign or malignant.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,10</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion, we present an unusual case of primary cutaneous CD4+ small- to medium-sized pleomorphic T-cell lymphoma with spontaneous remission. Because the clinical, histologic, and immunohistochemical characteristics of this type of lymphoma may coincide with those of other cutaneous lymphoid infiltrates, a careful study is required to reach a diagnosis. Dermatologists and pathologists should be aware of this entity in order to avoid unnecessary aggressive treatments.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Messeguer F, et al. Linfoma cutáneo primario de células T pleomórficas de pequeño y mediano CD4+: a propósito de un caso con resolución espontánea. Actas Dermosifiliogr.2011;102:636-8.</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" => 535 "Ancho" => 1472 "Tamanyo" => 146918 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Brownish-red skin tumor on the left cheek, with a smooth, shiny surface. B, Complete remission of the lesion 6 weeks later, showing residual hyperpigmentation and a depressed central area corresponding to the biopsy site.</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" => 714 "Ancho" => 952 "Tamanyo" => 273488 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Dense dermal infiltrate consisting of small- and medium-sized pleomorphic lymphocytes and histiocytes (hematoxylin–eosin, original magnification x250).</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" => 478 "Ancho" => 1676 "Tamanyo" => 231526 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A and B, Strongly positive staining for CD4 in almost the entire sample (original magnifications x20 and x100). 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 12 | 10 | 22 |
| 2024 Octubre | 76 | 35 | 111 |
| 2024 Septiembre | 75 | 26 | 101 |
| 2024 Agosto | 91 | 51 | 142 |
| 2024 Julio | 75 | 34 | 109 |
| 2024 Junio | 110 | 38 | 148 |
| 2024 Mayo | 85 | 35 | 120 |
| 2024 Abril | 89 | 27 | 116 |
| 2024 Marzo | 87 | 27 | 114 |
| 2024 Febrero | 61 | 20 | 81 |
| 2024 Enero | 64 | 27 | 91 |
| 2023 Diciembre | 62 | 15 | 77 |
| 2023 Noviembre | 72 | 30 | 102 |
| 2023 Octubre | 77 | 22 | 99 |
| 2023 Septiembre | 85 | 32 | 117 |
| 2023 Agosto | 49 | 22 | 71 |
| 2023 Julio | 59 | 43 | 102 |
| 2023 Junio | 63 | 21 | 84 |
| 2023 Mayo | 67 | 28 | 95 |
| 2023 Abril | 67 | 35 | 102 |
| 2023 Marzo | 54 | 21 | 75 |
| 2023 Febrero | 92 | 21 | 113 |
| 2023 Enero | 54 | 30 | 84 |
| 2022 Diciembre | 65 | 43 | 108 |
| 2022 Noviembre | 48 | 23 | 71 |
| 2022 Octubre | 37 | 32 | 69 |
| 2022 Septiembre | 33 | 35 | 68 |
| 2022 Agosto | 31 | 48 | 79 |
| 2022 Julio | 37 | 38 | 75 |
| 2022 Junio | 53 | 28 | 81 |
| 2022 Mayo | 39 | 56 | 95 |
| 2022 Abril | 78 | 46 | 124 |
| 2022 Marzo | 59 | 55 | 114 |
| 2022 Febrero | 51 | 27 | 78 |
| 2022 Enero | 65 | 45 | 110 |
| 2021 Diciembre | 41 | 41 | 82 |
| 2021 Noviembre | 53 | 45 | 98 |
| 2021 Octubre | 44 | 44 | 88 |
| 2021 Septiembre | 47 | 33 | 80 |
| 2021 Agosto | 47 | 41 | 88 |
| 2021 Julio | 58 | 27 | 85 |
| 2021 Junio | 70 | 32 | 102 |
| 2021 Mayo | 51 | 28 | 79 |
| 2021 Abril | 87 | 34 | 121 |
| 2021 Marzo | 70 | 18 | 88 |
| 2021 Febrero | 55 | 26 | 81 |
| 2021 Enero | 52 | 21 | 73 |
| 2020 Diciembre | 58 | 21 | 79 |
| 2020 Noviembre | 17 | 10 | 27 |
| 2020 Octubre | 19 | 12 | 31 |
| 2020 Septiembre | 38 | 11 | 49 |
| 2020 Agosto | 33 | 16 | 49 |
| 2020 Julio | 18 | 16 | 34 |
| 2020 Junio | 40 | 23 | 63 |
| 2020 Mayo | 25 | 17 | 42 |
| 2020 Abril | 25 | 18 | 43 |
| 2020 Marzo | 32 | 30 | 62 |
| 2020 Febrero | 3 | 7 | 10 |
| 2019 Diciembre | 4 | 5 | 9 |
| 2019 Noviembre | 0 | 2 | 2 |
| 2019 Octubre | 0 | 1 | 1 |
| 2019 Septiembre | 10 | 0 | 10 |
| 2019 Agosto | 0 | 3 | 3 |
| 2019 Julio | 0 | 12 | 12 |
| 2019 Junio | 0 | 31 | 31 |
| 2019 Mayo | 3 | 15 | 18 |
| 2019 Abril | 1 | 28 | 29 |
| 2019 Marzo | 0 | 9 | 9 |
| 2019 Febrero | 2 | 3 | 5 |
| 2019 Enero | 0 | 1 | 1 |
| 2018 Diciembre | 0 | 5 | 5 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Septiembre | 2 | 4 | 6 |
| 2018 Agosto | 0 | 2 | 2 |
| 2018 Julio | 0 | 3 | 3 |
| 2018 Junio | 0 | 1 | 1 |
| 2018 Mayo | 0 | 2 | 2 |
| 2018 Abril | 0 | 11 | 11 |
| 2018 Marzo | 3 | 0 | 3 |
| 2018 Febrero | 118 | 3 | 121 |
| 2018 Enero | 104 | 10 | 114 |
| 2017 Diciembre | 105 | 7 | 112 |
| 2017 Noviembre | 74 | 13 | 87 |
| 2017 Octubre | 84 | 7 | 91 |
| 2017 Septiembre | 123 | 12 | 135 |
| 2017 Agosto | 161 | 13 | 174 |
| 2017 Julio | 149 | 17 | 166 |
| 2017 Junio | 148 | 23 | 171 |
| 2017 Mayo | 138 | 9 | 147 |
| 2017 Abril | 140 | 19 | 159 |
| 2017 Marzo | 125 | 22 | 147 |
| 2017 Febrero | 125 | 13 | 138 |
| 2017 Enero | 76 | 15 | 91 |
| 2016 Diciembre | 114 | 10 | 124 |
| 2016 Noviembre | 158 | 10 | 168 |
| 2016 Octubre | 203 | 19 | 222 |
| 2016 Septiembre | 279 | 17 | 296 |
| 2016 Agosto | 150 | 9 | 159 |
| 2016 Julio | 112 | 15 | 127 |
| 2016 Junio | 11 | 10 | 21 |
| 2016 Mayo | 6 | 14 | 20 |
| 2016 Abril | 5 | 1 | 6 |
| 2016 Marzo | 14 | 8 | 22 |
| 2016 Febrero | 5 | 3 | 8 |
| 2016 Enero | 6 | 13 | 19 |
| 2015 Diciembre | 9 | 8 | 17 |
| 2015 Noviembre | 73 | 14 | 87 |
| 2015 Octubre | 56 | 1 | 57 |
| 2015 Septiembre | 20 | 1 | 21 |
| 2015 Agosto | 11 | 2 | 13 |
| 2015 Julio | 138 | 10 | 148 |
| 2015 Junio | 99 | 13 | 112 |
| 2015 Mayo | 124 | 16 | 140 |
| 2015 Abril | 122 | 11 | 133 |
| 2015 Marzo | 106 | 5 | 111 |
| 2015 Febrero | 119 | 15 | 134 |
| 2015 Enero | 106 | 14 | 120 |
| 2014 Diciembre | 95 | 13 | 108 |
| 2014 Noviembre | 85 | 13 | 98 |
| 2014 Octubre | 116 | 17 | 133 |
| 2014 Septiembre | 103 | 17 | 120 |
| 2014 Agosto | 119 | 14 | 133 |
| 2014 Julio | 96 | 23 | 119 |
| 2014 Junio | 168 | 12 | 180 |
| 2014 Mayo | 169 | 19 | 188 |
| 2014 Abril | 142 | 10 | 152 |
| 2014 Marzo | 163 | 21 | 184 |
| 2014 Febrero | 129 | 18 | 147 |
| 2014 Enero | 135 | 15 | 150 |
| 2013 Diciembre | 126 | 17 | 143 |
| 2013 Noviembre | 94 | 18 | 112 |
| 2013 Octubre | 68 | 17 | 85 |
| 2013 Septiembre | 65 | 12 | 77 |
| 2013 Agosto | 58 | 13 | 71 |
| 2013 Julio | 7 | 10 | 17 |
| 2013 Junio | 15 | 11 | 26 |
| 2013 Mayo | 16 | 14 | 30 |
| 2013 Abril | 15 | 21 | 36 |
| 2013 Marzo | 17 | 15 | 32 |
| 2013 Febrero | 35 | 9 | 44 |
| 2013 Enero | 19 | 7 | 26 |
| 2012 Diciembre | 18 | 4 | 22 |
| 2012 Noviembre | 0 | 4 | 4 |
| 2012 Octubre | 0 | 1 | 1 |
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