Case and Research Letters
A 65-Year-Old Woman With Multiple Papules in a Unilateral Segmental Distribution
Pápulas múltiples con distribución unilateral y segmentaria en mujer de 65 años
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Requena López, C. Maldonado Seral, B. Vivanco Allende" "autores" => array:3 [ 0 => array:4 [ "nombre" => "S." "apellidos" => "Requena López" "email" => array:1 [ 0 => "sheilarequenalopez@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" => "C." "apellidos" => "Maldonado Seral" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "B." "apellidos" => "Vivanco Allende" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Central de Asturias, Oviedo, Asturias, España" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario Central de Asturias, Oviedo, Asturias, España" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pápulas múltiples con distribución unilateral y segmentaria en mujer de 65 años" ] ] "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" => 2000 "Ancho" => 2677 "Tamanyo" => 995431 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Nests of basaloid cells among focal areas where strands of basaloid cells proliferate with pilar differentiation, scant mitotic activity, and a loose stroma. Hematoxylin-eosin.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Basaloid follicular hamartoma (BFH) is an uncommon benign neoplasm that may be familial or acquired, generalized or localized. Because the malignant transformation of this tumor to basal cell carcinoma has been reported, it is considered a premalignant lesion.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The characteristic histologic features of BFH are the proliferation of strands of basaloid epithelial cells that originate in the infundibular portion of hair follicles, branch outward, and are surrounded by a loose fibrous stroma. There is usually a clear transition between tumor cells and the adjacent stroma. The tumor cells do not usually display pleomorphism, nuclear atypia, or mitotic activity.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">BFH treatments include surgical excision, cryotherapy, laser therapy, topical imiquimod or retinoids, and photodynamic therapy.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">3,4</span></a> In case of malignant transformation to basal cell carcinoma, surgical removal is the treatment of choice because it has the lowest recurrence and complication rates.</p><p id="par0020" class="elsevierStylePara elsevierViewall">We report the case of a 60-year-old woman who complained of a pruritic lesion on her chest that had grown in recent months. She also reported having had many small lesions on the right side of her chest, pubic area and groin since the age of 30 years, although she never sought care because they were stable and asymptomatic. She did not report hair loss, ptosis, difficulty swallowing, or any other systemic symptoms, and she could not recall similar complaints in relatives. A physical examination revealed multiple skin-colored erythematous papules on the right side of her chest, right breast, pubic area, and groin. The lesions followed Blaschko lines, and some were comedones measuring less than 1<span class="elsevierStyleHsp" style=""></span>cm in diameter (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Above the right breast was an erythematous plaque measuring 1<span class="elsevierStyleHsp" style=""></span>cm. The surface was pearly and dermoscopy revealed telangiectases and blue-gray globules.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A biopsy of the supramammary plaque showed nests of basaloid cells that originated in the epidermal basement membrane; they were separated by fragments of healthy epidermis. The periphery was pale, and mitotic figures and apoptotic bodies were numerous. The lesion also contained areas where basaloid cells proliferated, arranged in strands with pilar differentiation, low mitotic activity, and a loose fibroblastic stroma (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">A diagnosis of basal cell carcinoma arising in a BFH was based on the clinical history, physical examination, and biopsy.</p><p id="par0035" class="elsevierStylePara elsevierViewall">This type of acquired, segmental, unilateral BFH can appear at birth or develop in adolescence. Unlike generalized familial forms, this phenotype is not associated with hypotrichosis, cystic fibrosis, or other autoimmune diseases such as myasthenia gravis or systemic lupus erythematosus.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">A diagnosis of BFH does not require laboratory tests or imaging. However, a complete medical history and physical examination is essential even though associations between localized BFH and systemic diseases have not been reported. If there is clinical suspicion of autoimmune disease, antinuclear antibody and antiacetylcholine receptor tests should be ordered in addition to any other tests the patient's symptoms suggest.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The main differential diagnoses are infundibulocystic basal cell carcinoma (IBCC)<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">2</span></a> and trichoepithelioma,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">5</span></a> which are also hair follicle tumors. BFH tends to center around the follicle and involve the interfollicular dermis less than IBCC. Deep infiltration, epidermal ulceration, and rapid growth would suggest IBCC. Some authors nevertheless consider BFH and IBCC to be the same diagnosis because their morphology is so similar and both express cytokeratin 20.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Trichoepithelioma can be distinguished from BFH by the greater stromal cellularity of the former. In addition, a trichoepithelioma shows fissures caused by retraction as the tumor stroma separates from the adjacent dermis.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Because of the coincidence of unilateral BFH and basal cell carcinoma lesions distributed along Blaschko lines in this case, segmental Gorlin syndrome had to be considered in the differential diagnosis.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> However, such a diagnosis was unlikely because of the absence of a family history of this syndrome or characteristic signs such as the presence of basal cell carcinoma from an early age, keratocystic odontogenic tumors in the maxilla, palmoplantar pitting, or developmental skeletal abnormalities. The unilateral distribution along Blaschko lines could be owing to the presence of a somatic mutation early in embryonic development.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Although detection of a mutation in the patched 1 gene (<span class="elsevierStyleItalic">PTCH1</span>) in healthy or affected skin,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a> or in peripheral blood lymphocytes, would have been useful for distinguishing BFH from segmental Gorlin syndrome in our patient, this gene test was unavailable in our hospital.</p><p id="par0065" class="elsevierStylePara elsevierViewall">In conclusion, although BFH is a very rare benign neoplasm, diagnosis is important so that periodic follow-up can be scheduled in the interest of early detection of basal cell carcinoma. BFH lesions can develop morphologic changes or grow and should be biopsied.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0070" 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" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Requena López S, Maldonado Seral C, Vivanco Allende B. Pápulas múltiples con distribución unilateral y segmentaria en mujer de 65 años. Actas Dermosifiliogr. 2018;109:190–192.</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" => 2000 "Ancho" => 2696 "Tamanyo" => 414731 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Multiple erythematous papules on the right chest and breast were distributed along Blaschko lines. The supramammary plaque (enlargement, upper right figure) had a pearly surface with telangiectases and globules. B, Papules and comedones in the right pubic area and groin.</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" => 2000 "Ancho" => 2677 "Tamanyo" => 995431 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Nests of basaloid cells among focal areas where strands of basaloid cells proliferate with pilar differentiation, scant mitotic activity, and a loose stroma. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 9 | 11 | 20 |
| 2024 Octubre | 80 | 49 | 129 |
| 2024 Septiembre | 72 | 24 | 96 |
| 2024 Agosto | 121 | 48 | 169 |
| 2024 Julio | 84 | 33 | 117 |
| 2024 Junio | 90 | 26 | 116 |
| 2024 Mayo | 84 | 36 | 120 |
| 2024 Abril | 75 | 33 | 108 |
| 2024 Marzo | 87 | 25 | 112 |
| 2024 Febrero | 73 | 35 | 108 |
| 2024 Enero | 60 | 31 | 91 |
| 2023 Diciembre | 85 | 26 | 111 |
| 2023 Noviembre | 89 | 30 | 119 |
| 2023 Octubre | 74 | 31 | 105 |
| 2023 Septiembre | 78 | 39 | 117 |
| 2023 Agosto | 60 | 19 | 79 |
| 2023 Julio | 81 | 23 | 104 |
| 2023 Junio | 82 | 20 | 102 |
| 2023 Mayo | 110 | 21 | 131 |
| 2023 Abril | 83 | 15 | 98 |
| 2023 Marzo | 80 | 20 | 100 |
| 2023 Febrero | 83 | 30 | 113 |
| 2023 Enero | 30 | 43 | 73 |
| 2022 Diciembre | 82 | 43 | 125 |
| 2022 Noviembre | 76 | 28 | 104 |
| 2022 Octubre | 69 | 21 | 90 |
| 2022 Septiembre | 115 | 48 | 163 |
| 2022 Agosto | 89 | 39 | 128 |
| 2022 Julio | 80 | 33 | 113 |
| 2022 Junio | 58 | 25 | 83 |
| 2022 Mayo | 136 | 32 | 168 |
| 2022 Abril | 145 | 43 | 188 |
| 2022 Marzo | 175 | 55 | 230 |
| 2022 Febrero | 179 | 27 | 206 |
| 2022 Enero | 224 | 34 | 258 |
| 2021 Diciembre | 135 | 42 | 177 |
| 2021 Noviembre | 127 | 48 | 175 |
| 2021 Octubre | 129 | 66 | 195 |
| 2021 Septiembre | 106 | 38 | 144 |
| 2021 Agosto | 169 | 29 | 198 |
| 2021 Julio | 157 | 29 | 186 |
| 2021 Junio | 185 | 47 | 232 |
| 2021 Mayo | 206 | 41 | 247 |
| 2021 Abril | 506 | 62 | 568 |
| 2021 Marzo | 313 | 34 | 347 |
| 2021 Febrero | 196 | 33 | 229 |
| 2021 Enero | 132 | 21 | 153 |
| 2020 Diciembre | 120 | 55 | 175 |
| 2020 Noviembre | 78 | 14 | 92 |
| 2020 Octubre | 72 | 15 | 87 |
| 2020 Septiembre | 58 | 15 | 73 |
| 2020 Agosto | 62 | 21 | 83 |
| 2020 Julio | 60 | 22 | 82 |
| 2020 Junio | 46 | 28 | 74 |
| 2020 Mayo | 30 | 10 | 40 |
| 2020 Abril | 30 | 11 | 41 |
| 2020 Marzo | 34 | 14 | 48 |
| 2020 Febrero | 11 | 0 | 11 |
| 2020 Enero | 4 | 0 | 4 |
| 2019 Diciembre | 8 | 0 | 8 |
| 2019 Noviembre | 2 | 0 | 2 |
| 2019 Septiembre | 10 | 0 | 10 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 8 | 0 | 8 |
| 2019 Abril | 2 | 0 | 2 |
| 2019 Marzo | 2 | 0 | 2 |
| 2019 Febrero | 2 | 0 | 2 |
| 2018 Diciembre | 3 | 0 | 3 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 2 | 0 | 2 |
| 2018 Abril | 2 | 0 | 2 |
| 2018 Febrero | 2 | 17 | 19 |
| 2018 Enero | 3 | 9 | 12 |
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