Case for Diagnosis
Hypopigmented Papules in a Healthy Girl
Pápulas hipopigmentadas en niña sana
C. López-Sánchez
, C. Downey, E. Baselga-Torres
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Servicio de Dermatología, Hospital de Sant Pau i la Santa Creu, Barcelona, España
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López-Sánchez, C. Downey, E. Baselga-Torres" "autores" => array:3 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "López-Sánchez" "email" => array:1 [ 0 => "clopezsan@santpau.cat" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Downey" ] 2 => array:2 [ "nombre" => "E." "apellidos" => "Baselga-Torres" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital de Sant Pau i la Santa Creu, Barcelona, España" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pápulas hipopigmentadas en niña sana" ] ] "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" => 748 "Ancho" => 1000 "Tamanyo" => 278417 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Hematoxylin-eosin,<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40. B) Periodic acid-Schiff,<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical History</span><p id="par0005" class="elsevierStylePara elsevierViewall">The patient was a 3-year-old girl with no history of interest who consulted for asymptomatic hypochromic lesions in the pubic region. The lesions had been present since birth, although they had gradually increased in number and size during the first months of life. Neither her parents nor her older brother had similar lesions.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">We observed minimally tender hypopigmented papules of different sizes clustered symmetrically in the pubic region (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). No lesions were observed in other areas.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">Histopathology was remarkable for the presence of a slightly acanthotic epidermis with mild orthokeratotic hyperkeratosis and numerous cells with a clear and abundant cytoplasm in the lower half of the epidermis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A). The clear cells were positive for periodic acid–Schiff (PAS) staining (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Additional Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Dermoscopy revealed well-defined hypopigmented papules measuring 3-4<span class="elsevierStyleHsp" style=""></span>mm with no desquamation or internal structures (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">What is your diagnosis?</p><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">Clear cell papulosis.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Given the benign nature of these lesions and the absence of symptoms, we recommended clinical observation.</p></span></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">Clear cell papulosis is an uncommon skin condition that affects children and appears as hypopigmented macules and papules generally on the lower abdomen and suprapubic area. Since it was first described in 1987 by Kuo et al.,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> 36 cases have been reported. Most involve patients of Asian origin and, to a lesser extent, patients of Hispanic or Indian origin, with a predominance in females (male:female, 1:1.4).<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">1–5</span></a> While the etiology of this condition is unknown, the fact that as many as one quarter of the cases reported occur in siblings has led some authors to favor a possible autosomal recessive inheritance pattern.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The lesions may be present at birth, although they usually develop during the first 2 years of life.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Clinically, they present as multiple, minimally palpable hypopigmented, nondesquamative macules or papules. The lesions are asymptomatic and are found mainly on the lower abdomen and pubis, although they have also been reported on the thorax, axillas, and limbs.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The differential diagnosis includes progressive macular hypomelanosis, verruca plana, pityriasis versicolor, guttate vitiligo, and nevus anemicus.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Histologically, the lesion is characterized by the presence of large cells with a benign appearance and abundant clear cytoplasm that occur in isolation or are found in small clusters along the basal layer and, occasionally, in the suprabasal layer of the epidermis. Other findings include mild hyperkeratosis, mild to moderate acanthosis, and reduced or no melanin in the basement membrane. The diagnosis is supported by positive staining of clear cells for mucin with PAS, mucicarmine, or alcian blue. Characteristic immunohistochemistry findings include strong positivity for cytokeratin AE1/AE3 and variable positivity for carcinoembryonic antigen and epithelial membrane antigen. Results are consistently negative for S100.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">It has traditionally been thought that there is a possible association between clear cells in clear cell papulosis and Toker cells, given the histological and immunohistochemical similarities between them and their distribution along the milk lines. The importance of this association lies in the potential role of Toker cells as precursors in mammary and extramammary Paget disease.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">4–6</span></a> However, current evidence is insufficient to support the hypothesis of an eventual transformation of clear cell papulosis to Paget disease.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3,5</span></a> On the contrary, long-term follow-up has revealed partial or complete resolution of the lesions in the vast majority of cases.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Recognition of this benign, asymptomatic lesion is important if we are to avoid unnecessary examinations and treatments.</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:7 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Clinical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Histopathology" ] 3 => array:3 [ "identificador" => "sec0020" "titulo" => "Additional Tests" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0025" "titulo" => "Diagnosis" ] 1 => array:2 [ "identificador" => "sec0030" "titulo" => "Clinical Course and Treatment" ] ] ] 4 => array:2 [ "identificador" => "sec0035" "titulo" => "Comment" ] 5 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflicts of Interest" ] 6 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: López-Sánchez C. Pápulas hipopigmentadas en niña sana Eczema y urticaria en Portugal. 2019;110:767–768.</p>" ] ] "multimedia" => array:3 [ 0 => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 680 "Ancho" => 905 "Tamanyo" => 53645 ] ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 748 "Ancho" => 1000 "Tamanyo" => 278417 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A) Hematoxylin-eosin,<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40. B) Periodic acid-Schiff,<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>40.</p>" ] ] 2 => array:6 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 788 "Ancho" => 905 "Tamanyo" => 61393 ] ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clear cell papulosis of the skin. A new entity with histogenetic implications for cutaneous Paget's disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "T.T. Kuo" 1 => "H.L. Chan" 2 => "S. 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Brown" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:4 [ "tituloSerie" => "Dermatol Online J" "fecha" => "2018" "volumen" => "24" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/29906016" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0060" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clear cell papulosis: A unique disorder in early childhood characterized by white macules in milk-line distribution" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "J.Y. Lee" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1046/j.1525-1470.1998.1998015328.x" "Revista" => array:6 [ "tituloSerie" => "Pediatr Dermatol" "fecha" => "1998" "volumen" => "15" "paginaInicial" => "328" "paginaFinal" => "329" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/9720709" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15782190/0000011000000009/v2_201911260739/S1578219019302677/v2_201911260739/en/main.assets" "Apartado" => array:4 [ "identificador" => "6154" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Case for Diagnosis" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15782190/0000011000000009/v2_201911260739/S1578219019302677/v2_201911260739/en/main.pdf?idApp=UINPBA000044&text.app=https://actasdermo.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219019302677?idApp=UINPBA000044" ]
| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 18 | 9 | 27 |
| 2024 Octubre | 114 | 54 | 168 |
| 2024 Septiembre | 121 | 31 | 152 |
| 2024 Agosto | 175 | 78 | 253 |
| 2024 Julio | 113 | 49 | 162 |
| 2024 Junio | 105 | 57 | 162 |
| 2024 Mayo | 106 | 54 | 160 |
| 2024 Abril | 89 | 39 | 128 |
| 2024 Marzo | 85 | 49 | 134 |
| 2024 Febrero | 89 | 36 | 125 |
| 2024 Enero | 80 | 37 | 117 |
| 2023 Diciembre | 84 | 26 | 110 |
| 2023 Noviembre | 99 | 52 | 151 |
| 2023 Octubre | 94 | 36 | 130 |
| 2023 Septiembre | 86 | 40 | 126 |
| 2023 Agosto | 72 | 31 | 103 |
| 2023 Julio | 125 | 49 | 174 |
| 2023 Junio | 70 | 39 | 109 |
| 2023 Mayo | 69 | 41 | 110 |
| 2023 Abril | 49 | 26 | 75 |
| 2023 Marzo | 78 | 29 | 107 |
| 2023 Febrero | 67 | 27 | 94 |
| 2023 Enero | 68 | 38 | 106 |
| 2022 Diciembre | 65 | 49 | 114 |
| 2022 Noviembre | 60 | 29 | 89 |
| 2022 Octubre | 44 | 27 | 71 |
| 2022 Septiembre | 69 | 65 | 134 |
| 2022 Agosto | 54 | 54 | 108 |
| 2022 Julio | 45 | 56 | 101 |
| 2022 Junio | 38 | 36 | 74 |
| 2022 Mayo | 102 | 43 | 145 |
| 2022 Abril | 83 | 28 | 111 |
| 2022 Marzo | 96 | 59 | 155 |
| 2022 Febrero | 80 | 18 | 98 |
| 2022 Enero | 112 | 41 | 153 |
| 2021 Diciembre | 88 | 41 | 129 |
| 2021 Noviembre | 88 | 38 | 126 |
| 2021 Octubre | 111 | 44 | 155 |
| 2021 Septiembre | 89 | 44 | 133 |
| 2021 Agosto | 133 | 38 | 171 |
| 2021 Julio | 97 | 25 | 122 |
| 2021 Junio | 79 | 33 | 112 |
| 2021 Mayo | 71 | 39 | 110 |
| 2021 Abril | 180 | 80 | 260 |
| 2021 Marzo | 97 | 29 | 126 |
| 2021 Febrero | 66 | 24 | 90 |
| 2021 Enero | 74 | 18 | 92 |
| 2020 Diciembre | 71 | 23 | 94 |
| 2020 Noviembre | 47 | 21 | 68 |
| 2020 Octubre | 46 | 24 | 70 |
| 2020 Septiembre | 32 | 23 | 55 |
| 2020 Agosto | 30 | 19 | 49 |
| 2020 Julio | 11 | 18 | 29 |
| 2020 Junio | 14 | 29 | 43 |
| 2020 Mayo | 14 | 10 | 24 |
| 2020 Abril | 15 | 9 | 24 |
| 2020 Marzo | 10 | 6 | 16 |
| 2020 Febrero | 3 | 2 | 5 |
| 2019 Noviembre | 8 | 0 | 8 |
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