Case and Research Letter
Inflammatory Disseminated Pruritic Porokeratosis With a Good Response to Ciclosporin
Poroqueratosis pruriginosa diseminada inflamatoria. Buena respuesta a ciclosporina
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Buena respuesta a ciclosporina" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "261" "paginaFinal" => "262" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Inflammatory Disseminated Pruritic Porokeratosis With a Good Response to Ciclosporin" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1597 "Ancho" => 1000 "Tamanyo" => 232332 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A-C) Múltiples pápulas eritemato-parduzcas distribuidas por el tronco y los miembros superiores e inferiores. 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Montes-Torres, C. Camarero-Mulas, D. de Argila, C. Gordillo, E. Daudén" "autores" => array:5 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Montes-Torres" "email" => array:1 [ 0 => "andreamtg@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" => "C." "apellidos" => "Camarero-Mulas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "D." "apellidos" => "de Argila" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "C." "apellidos" => "Gordillo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 4 => array:3 [ "nombre" => "E." "apellidos" => "Daudén" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario de La Princesa, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario de La Princesa, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Poroqueratosis pruriginosa diseminada inflamatoria. Buena respuesta a ciclosporina" ] ] "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" => 1597 "Ancho" => 1000 "Tamanyo" => 251948 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A-C, Multiple erythematous-brownish papules on the trunk and upper and lower extremities. D, Detail of a lesion on the left thigh, showing central atrophy with a well-defined, slightly elevated, keratotic border.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Six classic variants of porokeratosis (PK) have been described, only 3 of which (disseminated superficial PK [DSP], disseminated superficial actinic PK, and PK palmaris, plantaris et disseminata) are disseminated forms, characterized by a gradual onset and a mainly asymptomatic course.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a white man of 79 years of age with no past medical history of interest. He attended our clinic for the first time in February 2007 for multiple erythematous-brownish, slightly keratotic, and intensely pruritic papules that had arisen 2-and-a-half years earlier. The lesions had presented a rapid onset on the forearms and lower third of the legs, spreading to the limbs and trunk (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>); the number of lesions and the inflammatory activity had been variable. Successive skin biopsies revealed the presence of narrow columns of parakeratotic cells that crossed the stratum corneum, associated with an absence or reduction of the underlying stratum granulosum, marked spongiosis, and a dense superficial perivascular inflammatory infiltrate formed of lymphocytes and occasional eosinophils (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). In view of the intense pruritus, successive or combined treatments were tried with various topical agents, narrowband UV-B phototherapy, oral prednisone at different doses, acitretin, oral antihistamines, and serotonin reuptake inhibitors, none of which produced any significant improvement. In June 2011 the patient started treatment with oral ciclosporin, 4<span class="elsevierStyleHsp" style=""></span>mg/kg/d, which led to almost complete remission of his symptoms. Dose reductions below 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d or treatment interruption were always followed by an increase in the number and inflammatory activity of the lesions and an unrelenting pruritus very poorly tolerated by the patient. Treatment with ciclosporin has therefore been maintained at doses varying between 1 and 4<span class="elsevierStyleHsp" style=""></span>mg/kg/d, looking to achieve the minimal effective dose, with brief periods of treatment interruption, enabling the patient to maintain an adequate quality of life. No adverse effects have arisen to date, and blood tests and the blood pressure have remained within normal limits.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In 1992, Kanzaki et al.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> coined the term eruptive pruritic papular PK to refer to a previously undescribed condition of intensely pruritic monomorphic lesions of rapid onset in 3 Asian patients with a long-standing history of asymptomatic DSP. In 1995, a fourth case, identical to the previous ones, was published under the name of inflammatory disseminated superficial PK, after observing spongiosis and a dense lymphocytic inflammatory infiltrate on skin biopsy, supporting the hypothesis of an immune reaction against clones of mutated keratinocytes.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> The first case in a white patient was reported in 1997.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> That patient had no history of PK lesions and required systemic treatment to control the disease, contrasting with the spontaneous resolution observed in previous cases. A more recent publication described the case of an Asian man with a 13-year history of frequent periods of exacerbation.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> As in our case, no underlying disease was identified that could explain the persistent activity.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Recently, in order to reduce the conceptual confusion surrounding this rare variant, Shoimer et al.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> proposed unification of all the previous terminology under the name eruptive disseminated PK (EDP), to include all forms of rapid onset (less than 2 months), with multiple lesions and a histology compatible with PK. Additionally, they found that approximately 60% of cases were part of a paraneoplastic syndrome or were associated with immunosuppression. In our opinion, the term EDP is perhaps not the most appropriate. First, the mean clinical course of many previously published cases exceeds the proposed 2-month limit or was not even specified. And second, it should be recognized that the intense pruritus, present in almost all cases and frequently refractory to treatment,<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,5,6,8</span></a> is one of the condition's most notable traits. We therefore consider that the previously used terms “pruritic” and “inflammatory”, are more suitable, especially when the histopathology findings of this variant are also taken into account.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">With the exception of a limited number of cases that have required a period of systemic treatment,<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> the final tendency of the disease is towards spontaneous resolution within a few months to a year.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,4,8,10</span></a> No cases of malignant degeneration have been reported to date, though this has been observed in up to 10% of other variants of PK.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The typically limited clinical course of this disease would probably explain the absence of an association with malignant tumors. The persistent disease activity after 7 years of follow-up in our patient is of particular note, as is the good response to ciclosporin, a therapeutic option not previously described in the literature.</p><p id="par0030" class="elsevierStylePara elsevierViewall">We therefore propose the term “inflammatory disseminated pruritic porokeratosis” for this variant, and we have presented a case with frequent periods of exacerbation, unrelenting pruritus, and a good response to ciclosporin, drawing attention to the use of this therapeutic option in resistant cases.</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: Montes-Torres A, Camarero-Mulas C, de Argila D, Gordillo C, Daudén E. Poroqueratosis pruriginosa diseminada inflamatoria. Buena respuesta a ciclosporina. Actas Dermosifiliogr. 2016;107:261–262.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1597 "Ancho" => 1000 "Tamanyo" => 251948 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A-C, Multiple erythematous-brownish papules on the trunk and upper and lower extremities. D, Detail of a lesion on the left thigh, showing central atrophy with a well-defined, slightly elevated, keratotic border.</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" => 1268 "Ancho" => 1000 "Tamanyo" => 376843 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Cornoid lamella with intense underlying spongiosis and a dense superficial perivascular inflammatory infiltrate. Hematoxylin and eosin, original magnification ×100. B, Spongiosis and lymphocyte exocytosis in the epidermis beneath the cornoid lamella. There is an inflammatory infiltrate formed mainly of lymphocytes, with occasional eosinophils. Hematoxylin and eosin, original magnification ×200.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Porokeratosis: Present concepts" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "P. Sertznig" 1 => "V. von Felbert" 2 => "M. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 6 | 5 | 11 |
| 2024 Octubre | 68 | 44 | 112 |
| 2024 Septiembre | 75 | 25 | 100 |
| 2024 Agosto | 95 | 53 | 148 |
| 2024 Julio | 75 | 38 | 113 |
| 2024 Junio | 96 | 58 | 154 |
| 2024 Mayo | 62 | 32 | 94 |
| 2024 Abril | 67 | 35 | 102 |
| 2024 Marzo | 70 | 34 | 104 |
| 2024 Febrero | 49 | 39 | 88 |
| 2024 Enero | 46 | 26 | 72 |
| 2023 Diciembre | 38 | 21 | 59 |
| 2023 Noviembre | 65 | 25 | 90 |
| 2023 Octubre | 58 | 26 | 84 |
| 2023 Septiembre | 72 | 33 | 105 |
| 2023 Agosto | 52 | 22 | 74 |
| 2023 Julio | 64 | 38 | 102 |
| 2023 Junio | 52 | 19 | 71 |
| 2023 Mayo | 43 | 22 | 65 |
| 2023 Abril | 51 | 32 | 83 |
| 2023 Marzo | 31 | 25 | 56 |
| 2023 Febrero | 36 | 16 | 52 |
| 2023 Enero | 31 | 36 | 67 |
| 2022 Diciembre | 56 | 35 | 91 |
| 2022 Noviembre | 30 | 21 | 51 |
| 2022 Octubre | 28 | 16 | 44 |
| 2022 Septiembre | 16 | 30 | 46 |
| 2022 Agosto | 20 | 33 | 53 |
| 2022 Julio | 25 | 26 | 51 |
| 2022 Junio | 15 | 22 | 37 |
| 2022 Mayo | 19 | 23 | 42 |
| 2022 Abril | 25 | 24 | 49 |
| 2022 Marzo | 34 | 33 | 67 |
| 2022 Febrero | 30 | 34 | 64 |
| 2022 Enero | 19 | 33 | 52 |
| 2021 Diciembre | 18 | 46 | 64 |
| 2021 Noviembre | 35 | 38 | 73 |
| 2021 Octubre | 30 | 49 | 79 |
| 2021 Septiembre | 30 | 38 | 68 |
| 2021 Agosto | 34 | 23 | 57 |
| 2021 Julio | 36 | 24 | 60 |
| 2021 Junio | 20 | 24 | 44 |
| 2021 Mayo | 22 | 30 | 52 |
| 2021 Abril | 44 | 33 | 77 |
| 2021 Marzo | 45 | 27 | 72 |
| 2021 Febrero | 44 | 22 | 66 |
| 2021 Enero | 32 | 19 | 51 |
| 2020 Diciembre | 18 | 15 | 33 |
| 2020 Noviembre | 33 | 24 | 57 |
| 2020 Octubre | 15 | 16 | 31 |
| 2020 Septiembre | 26 | 12 | 38 |
| 2020 Agosto | 18 | 17 | 35 |
| 2020 Julio | 12 | 13 | 25 |
| 2020 Junio | 32 | 24 | 56 |
| 2020 Mayo | 25 | 18 | 43 |
| 2020 Abril | 23 | 14 | 37 |
| 2020 Marzo | 32 | 13 | 45 |
| 2020 Febrero | 2 | 0 | 2 |
| 2020 Enero | 0 | 1 | 1 |
| 2019 Diciembre | 0 | 2 | 2 |
| 2019 Noviembre | 0 | 4 | 4 |
| 2019 Septiembre | 0 | 4 | 4 |
| 2019 Agosto | 0 | 1 | 1 |
| 2019 Julio | 0 | 1 | 1 |
| 2019 Junio | 2 | 14 | 16 |
| 2019 Mayo | 2 | 25 | 27 |
| 2019 Abril | 13 | 1 | 14 |
| 2019 Marzo | 1 | 3 | 4 |
| 2019 Febrero | 1 | 0 | 1 |
| 2019 Enero | 8 | 0 | 8 |
| 2018 Diciembre | 5 | 0 | 5 |
| 2018 Noviembre | 12 | 0 | 12 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 5 | 0 | 5 |
| 2018 Febrero | 43 | 1 | 44 |
| 2018 Enero | 52 | 8 | 60 |
| 2017 Diciembre | 68 | 9 | 77 |
| 2017 Noviembre | 22 | 4 | 26 |
| 2017 Octubre | 24 | 7 | 31 |
| 2017 Septiembre | 12 | 9 | 21 |
| 2017 Agosto | 24 | 5 | 29 |
| 2017 Julio | 15 | 10 | 25 |
| 2017 Junio | 30 | 7 | 37 |
| 2017 Mayo | 24 | 8 | 32 |
| 2017 Abril | 22 | 7 | 29 |
| 2017 Marzo | 11 | 4 | 15 |
| 2017 Febrero | 14 | 8 | 22 |
| 2017 Enero | 21 | 7 | 28 |
| 2016 Diciembre | 30 | 10 | 40 |
| 2016 Noviembre | 17 | 18 | 35 |
| 2016 Octubre | 15 | 18 | 33 |
| 2016 Septiembre | 1 | 0 | 1 |
| 2016 Agosto | 1 | 1 | 2 |
| 2016 Julio | 1 | 0 | 1 |
| 2016 Junio | 0 | 2 | 2 |
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