Case and Research Letter
A Simple and Effective Method for Treating Cylindromas in Brooke-Spiegler Syndrome
Método simple y efectivo para el tratamiento de los cilindromas en el síndrome de Brooke-Spiegler
S. Lucas-Truyols
, C. Lloret-Ruiz, F. Millán-Parrilla, E. Gimeno-Carpio
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Servicio de Dermatología, Hospital Arnau de Vilanova, Valencia, Spain
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Lucas-Truyols, C. Lloret-Ruiz, F. Millán-Parrilla, E. Gimeno-Carpio" "autores" => array:4 [ 0 => array:4 [ "nombre" => "S." "apellidos" => "Lucas-Truyols" "email" => array:1 [ 0 => "sofialucas89@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Lloret-Ruiz" ] 2 => array:2 [ "nombre" => "F." "apellidos" => "Millán-Parrilla" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Gimeno-Carpio" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Arnau de Vilanova, Valencia, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Método simple y efectivo para el tratamiento de los cilindromas en el síndrome de Brooke-Spiegler" ] ] "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" => 1084 "Ancho" => 1499 "Tamanyo" => 573179 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cylindroma on the scalp.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The genetic skin disease known as Brooke-Spiegler syndrome (BSS) is a rare autosomal dominant condition with more than 50 cases reported to date.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> BSS predisposes to benign adnexal tumors that generally first appear in adulthood and involve the folliculo-sebaceous-apocrine unit. It is characterized by multiple cylindromas, trichoepitheliomas, and, occasionally, spiradenomas.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The tumors appear simultaneously, as in BSS, or in isolation, as in familial cylindromatosis and multiple familial trichoepithelioma. Consequently, they seem to have a common origin in a pluripotent cell in the folliculo-sebaceous-apocrine unit, with several phenotypic variants.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Various mutations have been identified in the tumor suppressor gene <span class="elsevierStyleItalic">CYLD</span> on chromosome 16q12-913, thus indicating an association with BSS. <span class="elsevierStyleItalic">CYLD</span> inhibits the tumor necrosis factor α pathway, diminishing expression of nuclear factor κB (NF-κB), a transcription factor that in turn regulates various antiapoptotic genes involved in the proliferation of adnexa. A loss of the suppressive function of <span class="elsevierStyleItalic">CYLD</span> leads to greater resistance to apoptosis and, consequently, the appearance of these tumors.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3–5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Clinically, cylindromas present as pink nodules on the scalp and behind the ears (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Trichoepitheliomas appear as papules or small translucent or flesh-colored nodules and are located mainly in the center of the face, especially in the nasal region. Spiradenomas, on the other hand, usually present as solitary bluish or flesh-colored nodules that are often painful.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The emotional impact of recurrence of these lesions on the face and scalp makes treatment essential. Our literature search revealed various options, such as electrocautery, dermabrasion, radiofrequency, cryotherapy, CO<span class="elsevierStyleInf">2</span> laser,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> radiotherapy (in special cases),<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> and, recently, enucleation.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> The treatment recommended for solitary cylindromas is surgical removal, whereas partial or total skin grafting has been proposed for multiple lesions.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> NF-κB inhibitors, such as acetylsalicylic acid and its derivatives, may prove useful both for prevention and for treatment of cylindromas when applied locally.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Therefore, treatment should be tailored depending on the patient, size of the lesion, number of lesions, therapeutic options available, and the dermatologist's experience.</p><p id="par0025" class="elsevierStylePara elsevierViewall">We report the case of an 80-year-old woman with BSS who presented with multiple cylindromas on the scalp that had recurred since adolescence, together with occasional spiradenomas. Her family history was remarkable in that one of her daughters, aged 49 years, had had numerous facial trichoepitheliomas since puberty. She has been followed regularly in our department for the last 10 years, and several lesions have been removed using elliptical excision and direct closure. Given the high number of lesions and the excisions performed, we sought an alternative that would make for more efficient surgery and benefit the patient by reducing the number of trips to the hospital. Five years ago, we began to perform a technique that combined cytoreduction and subsequent curettage of the growths. Our technique is minimally invasive and efficacious.</p><p id="par0030" class="elsevierStylePara elsevierViewall">After administration of local anesthetic (mepivacaine 2%), an incision is made with a scalpel or scissor cut to enable resection of most of the tumor mass and create a portal of entry for the next step. After debulking, the remaining lesional tissue is removed by intense curettage, and bleeding is stopped using electrocautery. The resulting wound heals by secondary intention (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Complete scarring is achieved within approximately 14 days based on daily dressing with antiseptic and antibiotic ointment. The wound progresses without complications after surgery, and the cosmetic outcome is favorable at the 12-month visit (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Regular follow-up has revealed a reduction in the number of cylindromas and a rate of recurrence of the lesions removed lower than 25%. When the lesion did relapse, the growth was always smaller than the original lesion, thus facilitating surgery.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Our technique enables us to reduce the volume of the lesions by eliminating several growths during the same procedure. This is particularly advantageous in patients who must undergo repeated procedures. It also enables us to concentrate on the emergence of new lesions, since cylindromas rarely recur after removal using this technique. This is because elimination of the lesions by combining several techniques in one procedure (eg, debulking with scissors, followed by curettage of the wound bed, and subsequent use of an electric scalpel) enables almost total necrosis of any remaining cells and destruction of the hair follicles, which are a potential source of new tumors.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0040" 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: Lucas-Truyols S, Lloret-Ruiz C, Millán-Parrilla F, Gimeno-Carpio E. Método simple y efectivo para el tratamiento de los cilindromas en el síndrome de Brooke-Spiegler. Actas Dermosifiliogr. 2017;108:692–694.</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" => 1084 "Ancho" => 1499 "Tamanyo" => 573179 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cylindroma on the scalp.</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" => 1277 "Ancho" => 1700 "Tamanyo" => 588058 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Incision and debulking. B, Curettage. C, Electrocautery to stop bleeding. D, Immediate postoperative findings.</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" => 1488 "Ancho" => 1500 "Tamanyo" => 661024 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Outcome 12 months after surgery.</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" => "Heterozygous cylindromatosis gene mutation c.1628_1629delct in a family with Brook-Spiegler syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "C.A. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 21 | 13 | 34 |
| 2024 Octubre | 193 | 79 | 272 |
| 2024 Septiembre | 214 | 53 | 267 |
| 2024 Agosto | 264 | 69 | 333 |
| 2024 Julio | 252 | 55 | 307 |
| 2024 Junio | 195 | 53 | 248 |
| 2024 Mayo | 178 | 66 | 244 |
| 2024 Abril | 176 | 32 | 208 |
| 2024 Marzo | 240 | 40 | 280 |
| 2024 Febrero | 202 | 48 | 250 |
| 2024 Enero | 185 | 31 | 216 |
| 2023 Diciembre | 236 | 20 | 256 |
| 2023 Noviembre | 252 | 36 | 288 |
| 2023 Octubre | 257 | 25 | 282 |
| 2023 Septiembre | 233 | 32 | 265 |
| 2023 Agosto | 256 | 20 | 276 |
| 2023 Julio | 216 | 52 | 268 |
| 2023 Junio | 212 | 38 | 250 |
| 2023 Mayo | 272 | 33 | 305 |
| 2023 Abril | 206 | 30 | 236 |
| 2023 Marzo | 184 | 28 | 212 |
| 2023 Febrero | 161 | 28 | 189 |
| 2023 Enero | 130 | 71 | 201 |
| 2022 Diciembre | 141 | 53 | 194 |
| 2022 Noviembre | 101 | 39 | 140 |
| 2022 Octubre | 73 | 28 | 101 |
| 2022 Septiembre | 43 | 58 | 101 |
| 2022 Agosto | 32 | 49 | 81 |
| 2022 Julio | 112 | 46 | 158 |
| 2022 Junio | 43 | 42 | 85 |
| 2022 Mayo | 201 | 51 | 252 |
| 2022 Abril | 295 | 34 | 329 |
| 2022 Marzo | 279 | 53 | 332 |
| 2022 Febrero | 262 | 39 | 301 |
| 2022 Enero | 272 | 34 | 306 |
| 2021 Diciembre | 204 | 43 | 247 |
| 2021 Noviembre | 175 | 45 | 220 |
| 2021 Octubre | 182 | 53 | 235 |
| 2021 Septiembre | 246 | 42 | 288 |
| 2021 Agosto | 190 | 43 | 233 |
| 2021 Julio | 165 | 32 | 197 |
| 2021 Junio | 163 | 25 | 188 |
| 2021 Mayo | 164 | 54 | 218 |
| 2021 Abril | 474 | 79 | 553 |
| 2021 Marzo | 210 | 24 | 234 |
| 2021 Febrero | 156 | 37 | 193 |
| 2021 Enero | 124 | 18 | 142 |
| 2020 Diciembre | 95 | 20 | 115 |
| 2020 Noviembre | 79 | 25 | 104 |
| 2020 Octubre | 76 | 20 | 96 |
| 2020 Septiembre | 72 | 21 | 93 |
| 2020 Agosto | 50 | 22 | 72 |
| 2020 Julio | 27 | 16 | 43 |
| 2020 Junio | 51 | 30 | 81 |
| 2020 Mayo | 21 | 15 | 36 |
| 2020 Abril | 32 | 9 | 41 |
| 2020 Marzo | 19 | 19 | 38 |
| 2020 Febrero | 4 | 0 | 4 |
| 2020 Enero | 2 | 0 | 2 |
| 2019 Diciembre | 6 | 0 | 6 |
| 2019 Noviembre | 2 | 0 | 2 |
| 2019 Septiembre | 9 | 0 | 9 |
| 2019 Agosto | 2 | 0 | 2 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 3 | 0 | 3 |
| 2019 Abril | 11 | 0 | 11 |
| 2019 Marzo | 3 | 0 | 3 |
| 2019 Febrero | 5 | 0 | 5 |
| 2019 Enero | 4 | 0 | 4 |
| 2018 Diciembre | 13 | 0 | 13 |
| 2018 Octubre | 6 | 0 | 6 |
| 2018 Septiembre | 5 | 0 | 5 |
| 2018 Febrero | 30 | 12 | 42 |
| 2018 Enero | 60 | 8 | 68 |
| 2017 Diciembre | 62 | 7 | 69 |
| 2017 Noviembre | 76 | 8 | 84 |
| 2017 Octubre | 58 | 19 | 77 |
| 2017 Septiembre | 232 | 30 | 262 |
| 2017 Agosto | 14 | 17 | 31 |
| 2017 Julio | 5 | 13 | 18 |
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