Case and Research Letters
Why Is Mohs Micrographic Surgery Underused in the Treatment of Dermatofibrosarcoma Protuberans in Children?
¿Por qué la cirugía micrográfica de Mohs está infrautilizada en el tratamiento del dermatofibrosarcoma protuberans infantil?
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Proliferación de células fusiformes organizadas en fascículos (hematoxilina-eosina<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100). B. Espacios pseudovasculares irregulares delineados por células gigantes multinucleadas (hematoxilina-eosina<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100) C. Ocupación de la dermis con extensión al tejido celular subcutáneo (hematoxilina-eosina<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20) D. Las células fusiformes expresan el marcador CD34 (×<span class="elsevierStyleHsp" style=""></span>20).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "E. Jubert, L.J. del Pozo, C. Saus, A. Martín-Santiago" "autores" => array:4 [ 0 => array:2 [ "nombre" => "E." "apellidos" => "Jubert" ] 1 => array:2 [ "nombre" => "L.J." "apellidos" => "del Pozo" ] 2 => array:2 [ "nombre" => "C." 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"apellidos" => "Bassas-Vila" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001731015003476" "doi" => "10.1016/j.ad.2015.07.009" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731015003476?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219016000123?idApp=UINPBA000044" "url" => "/15782190/0000010700000002/v2_201703220318/S1578219016000123/v2_201703220318/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letters</span>" "titulo" => "Why Is Mohs Micrographic Surgery Underused in the Treatment of Dermatofibrosarcoma Protuberans in Children?" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">To the Editor:</span>" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "158" "paginaFinal" => "162" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E. Jubert, L.J. del Pozo, C. Saus, A. Martín-Santiago" "autores" => array:4 [ 0 => array:3 [ "nombre" => "E." "apellidos" => "Jubert" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "L.J." "apellidos" => "del Pozo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Saus" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:4 [ "nombre" => "A." "apellidos" => "Martín-Santiago" "email" => array:1 [ 0 => "anahsd@telefonica.net" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Son Espases, Palma de Mallorca, España" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario Son Espases, Palma de Mallorca, España" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "¿Por qué la cirugía micrográfica de Mohs está infrautilizada en el tratamiento del dermatofibrosarcoma protuberans infantil?" ] ] "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" => 1915 "Ancho" => 1800 "Tamanyo" => 932647 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A. Proliferation of spindle cells arranged in fascicles (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100). B, Irregular pseudovascular spaces surrounded by multinucleated giant cells (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100).<span class="elsevierStyleHsp" style=""></span>C, Involvement of full thickness of the dermis with extension into the subcutaneous tissue (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20). D, Spindle cells expressing CD34 (×<span class="elsevierStyleHsp" style=""></span>20).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Dermatofibrosarcoma protuberans (DFSP) is a slow-growing, intermediate-grade fibrohistiocytic tumor characterized by high rates of local recurrence but low metastatic potential. Few cases of congenital forms have been reported, but this is probably because early lesions can go unnoticed or be confused with other entities. There is evidence that recurrence rates are lower with Mohs micrographic surgery (MMS) than with conventional surgery with wide margins.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> Nonetheless, in 3 recently published pediatric series, wide excision was used in the vast majority of patients (41/43).<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">2–4</span></a> We present a case of congenital giant-cell fibroblastoma, a histologic subtype of DFSP, which was diagnosed and treated in an 8-year-old boy. In this report, we highlight the advantages of MMS and the use of negative-pressure wound therapy (NPWT) in children with DFSP.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was an 8-year-old boy with no relevant past history who was seen for a lesion in the right axilla that had grown and become progressively harder. The lesion, which was initially flat and reddish in color, had been present since birth. Examination revealed an indurated, mobile plaque measuring 4<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2.5<span class="elsevierStyleHsp" style=""></span>cm. The plaque was pink but had a purple, slightly elevated area. The regional lymph nodes were not enlarged. Biopsy showed a poorly delimited tumorous lesion composed of spindle and oval cells that occupied practically the entire dermis and extended down into the subcutaneous tissue. Pseudovascular spaces surrounded by multinucleated giant cells were also visible. The spindle cells expressed CD34 and were negative for S-100 protein and smooth muscle actin (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A diagnosis of giant cell fibroblastoma was established, and extracutaneous invasion was ruled out by magnetic resonance imaging. The tumor was excised under general anesthesia. The technique chosen was slow MMS (Breuninger technique), with excision of 1-cm margins in the first stage (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). This technique consists of the en bloc resection of the tumor at a 90° angle to the surface and the achievement of a uniform wound bed. In our case, the depth of excision reached the fascia. The resected specimen was fixed in formol and embedded in paraffin. Because the slides with samples from the deep and peripheral margins were to be reviewed at a later date, it was decided to cover the surgical defect with a NPWT system with a negative continuous pressure of 125 mm<span class="elsevierStyleHsp" style=""></span>Hg (Kinetic Concepts Inc.) (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a> A-C). On day 9, with histologic confirmation of tumor-free margins, the surgical defect was reconstructed using a Limberg flap over the granulation tissue formed during NPWT; no specific preparation was required (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>D). There have been no recurrences in 4 years of follow-up.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">DFSP is a rare tumor in children, with an estimated incidence of 1 case per 1 000 000 population under the age of 20 years.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> Its prevalence could, however, be underestimated as time to diagnosis ranges from 5 to 15 years. Congenital forms of DFSP are even rarer. In 1 series of 152 cases of childhood DFSP, for example, only 20 tumors were congenital.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Childhood and adult forms are similar in terms of immunohistochemical characteristics, the translocation of genetic material between chromosomes 17 and 22, and the association with the <span class="elsevierStyleItalic">COL1A1-PDGFB</span> fusion gene. Congenital variants of DFSP present more frequently as a stain or an atrophic plaque than as a tumorous lesion. The differential diagnosis is broad and includes vascular anomalies, aplasia cutis congenita, atrophoderma, myofibromatosis, childhood fibromatosis, and fibrosarcomas.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Compared with adults, in children, tumors are more frequently located on the lower extremities and at acral sites. Other differences in children include lower recurrence rates, very few cases of metastasis, and little use of MMS.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The treatment of choice for DFSP is surgery,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">7</span></a> with imatinib<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">8</span></a> and radiation therapy reserved for recurrent, unresectable, or metastatic tumors. In a report of 74 patients, mostly adults, with DFSP, and a review of the literature, Serra-Guillen et al.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> concluded that MMS was associated with considerably fewer recurrences than conventional surgery with wide margins, and also achieved greater preservation of healthy tissue. The most common MMS technique used is slow MMS, which requires coverage of the surgical defect with biosynthetic skin substitutes or other dressings. NPWT, however, has been proposed as an alternative to skin substitutes for temporary wound coverage.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a> This system reduces the need for analgesia and sedation and also requires less frequent wound dressing. In our case, it additionally allowed the patient to walk around and move his arm. Portable vacuum systems can be used on an outpatient basis, but because of the geographic distribution of our community (our hospital is on an island), we were not able to use a portable NPWT system.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In 2014, there were 3 reports of childhood DFSP in hospitals in Taiwan,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> Canada,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> and Spain.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a> The series included, respectively, 13, 17, and 13 patients under the age of 18 years, but surprisingly, MMS was used in just 2 of these 43 patients. If MMS has been shown to achieve better therapeutic, functional, and cosmetic results, why is it underused in children with DFSP? Finally, we would like to stress the importance of working closely with multidisciplinary teams to ensure that children with DFSP are diagnosed and treated correctly.</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: Jubert E, del Pozo LJ, Saus C, Martín-Santiago A. ¿Por qué la cirugía micrográfica de Mohs está infrautilizada en el tratamiento del dermatofibrosarcoma protuberans infantil?. Actas Dermosifiliogr. 2016;107:158–162.</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" => 1915 "Ancho" => 1800 "Tamanyo" => 932647 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A. Proliferation of spindle cells arranged in fascicles (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100). B, Irregular pseudovascular spaces surrounded by multinucleated giant cells (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>100).<span class="elsevierStyleHsp" style=""></span>C, Involvement of full thickness of the dermis with extension into the subcutaneous tissue (original magnification, hematoxylin-eosin<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>20). D, Spindle cells expressing CD34 (×<span class="elsevierStyleHsp" style=""></span>20).</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" => 1872 "Ancho" => 1500 "Tamanyo" => 573330 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Schematic of specimen. B, Ring-shaped excision of a 1-cm margin of healthy skin around the debulking specimen. C, Specimen from first slow MMS stage embedded in paraffin for examination. D, Final surgical defect measuring 6<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>4.5<span class="elsevierStyleHsp" style=""></span>cm.</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" => 1587 "Ancho" => 1900 "Tamanyo" => 531959 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Negative pressure wound therapy (NPWT) system. A, Coverage of skin defect with foam. B, Placement of adhesive film over the foam to seal the system. C, Placement of vacuum tube connected to NPWT system. D, Reconstruction of surgical defect with Limberg flap.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Mohs micrographic surgery in dermatofibrosarcoma protuberans allows tumour clearance with smaller margins and greater preservation of healthy tissue compared with conventional surgery: A study of 74 primary cases" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "C. Serra-Guillén" 1 => "B. Llombart" 2 => "E. Nagore" 3 => "C. Guillén" 4 => "C. Requena" 5 => "V. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 4 | 0 | 4 |
| 2024 Octubre | 74 | 36 | 110 |
| 2024 Septiembre | 81 | 18 | 99 |
| 2024 Agosto | 90 | 59 | 149 |
| 2024 Julio | 71 | 35 | 106 |
| 2024 Junio | 67 | 33 | 100 |
| 2024 Mayo | 78 | 37 | 115 |
| 2024 Abril | 68 | 21 | 89 |
| 2024 Marzo | 67 | 28 | 95 |
| 2024 Febrero | 64 | 37 | 101 |
| 2024 Enero | 58 | 33 | 91 |
| 2023 Diciembre | 74 | 23 | 97 |
| 2023 Noviembre | 72 | 24 | 96 |
| 2023 Octubre | 82 | 23 | 105 |
| 2023 Septiembre | 86 | 40 | 126 |
| 2023 Agosto | 75 | 10 | 85 |
| 2023 Julio | 60 | 33 | 93 |
| 2023 Junio | 54 | 31 | 85 |
| 2023 Mayo | 50 | 21 | 71 |
| 2023 Abril | 48 | 20 | 68 |
| 2023 Marzo | 54 | 22 | 76 |
| 2023 Febrero | 58 | 37 | 95 |
| 2023 Enero | 35 | 25 | 60 |
| 2022 Diciembre | 52 | 34 | 86 |
| 2022 Noviembre | 31 | 17 | 48 |
| 2022 Octubre | 30 | 25 | 55 |
| 2022 Septiembre | 22 | 38 | 60 |
| 2022 Agosto | 19 | 33 | 52 |
| 2022 Julio | 26 | 34 | 60 |
| 2022 Junio | 18 | 22 | 40 |
| 2022 Mayo | 33 | 33 | 66 |
| 2022 Abril | 59 | 28 | 87 |
| 2022 Marzo | 41 | 42 | 83 |
| 2022 Febrero | 30 | 20 | 50 |
| 2022 Enero | 40 | 23 | 63 |
| 2021 Diciembre | 33 | 26 | 59 |
| 2021 Noviembre | 43 | 41 | 84 |
| 2021 Octubre | 54 | 42 | 96 |
| 2021 Septiembre | 41 | 32 | 73 |
| 2021 Agosto | 48 | 24 | 72 |
| 2021 Julio | 48 | 24 | 72 |
| 2021 Junio | 48 | 37 | 85 |
| 2021 Mayo | 75 | 21 | 96 |
| 2021 Abril | 61 | 39 | 100 |
| 2021 Marzo | 76 | 15 | 91 |
| 2021 Febrero | 56 | 25 | 81 |
| 2021 Enero | 42 | 15 | 57 |
| 2020 Diciembre | 48 | 19 | 67 |
| 2020 Noviembre | 28 | 17 | 45 |
| 2020 Octubre | 35 | 12 | 47 |
| 2020 Septiembre | 36 | 8 | 44 |
| 2020 Agosto | 28 | 26 | 54 |
| 2020 Julio | 19 | 24 | 43 |
| 2020 Junio | 48 | 31 | 79 |
| 2020 Mayo | 26 | 20 | 46 |
| 2020 Abril | 37 | 20 | 57 |
| 2020 Marzo | 28 | 15 | 43 |
| 2020 Febrero | 7 | 3 | 10 |
| 2019 Diciembre | 4 | 2 | 6 |
| 2019 Septiembre | 3 | 0 | 3 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Mayo | 3 | 1 | 4 |
| 2019 Abril | 0 | 1 | 1 |
| 2019 Marzo | 0 | 5 | 5 |
| 2019 Febrero | 4 | 0 | 4 |
| 2018 Diciembre | 2 | 0 | 2 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 5 | 0 | 5 |
| 2018 Agosto | 0 | 2 | 2 |
| 2018 Mayo | 0 | 8 | 8 |
| 2018 Marzo | 1 | 0 | 1 |
| 2018 Febrero | 23 | 5 | 28 |
| 2018 Enero | 31 | 4 | 35 |
| 2017 Diciembre | 27 | 8 | 35 |
| 2017 Noviembre | 41 | 8 | 49 |
| 2017 Octubre | 23 | 6 | 29 |
| 2017 Septiembre | 13 | 6 | 19 |
| 2017 Agosto | 19 | 8 | 27 |
| 2017 Julio | 26 | 7 | 33 |
| 2017 Junio | 24 | 7 | 31 |
| 2017 Mayo | 19 | 15 | 34 |
| 2017 Abril | 19 | 3 | 22 |
| 2017 Marzo | 18 | 38 | 56 |
| 2017 Febrero | 12 | 4 | 16 |
| 2017 Enero | 21 | 7 | 28 |
| 2016 Diciembre | 25 | 12 | 37 |
| 2016 Noviembre | 18 | 10 | 28 |
| 2016 Octubre | 17 | 17 | 34 |
| 2016 Junio | 0 | 4 | 4 |
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