Case and Research Letter
Pediatric generalized morphea that developed at a BCG vaccination site
Morfea generalizada pediátrica de aparición en el sitio de la inyección de la vacuna BCG
Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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(A) Normal epidermis with no inflammatory infiltrate. Miniaturized hair follicles (red arrow), atrophy of the sebaceous glands (yellow arrow) (hematoxylin eosin, original magnification 100×). (B) Normal dermal elastic fibers (orcein, original magnification 40×). (C) Perifollicular fibrosis (trichome, original magnification 100×).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. César, T. Baudrier, A. Mota, F. Azevedo" "autores" => array:4 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "César" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Baudrier" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Mota" ] 3 => array:2 [ "nombre" => "F." "apellidos" => "Azevedo" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S0001731014004116" "doi" => "10.1016/j.ad.2014.08.008" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731014004116?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219015000335?idApp=UINPBA000044" "url" => "/15782190/0000010600000002/v2_201502240233/S1578219015000335/v2_201502240233/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "Pediatric generalized morphea that developed at a BCG vaccination site" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "150" "paginaFinal" => "152" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M. Matsumoto, T. Yamamoto" "autores" => array:2 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Matsumoto" ] 1 => array:4 [ "nombre" => "T." "apellidos" => "Yamamoto" "email" => array:1 [ 0 => "toyamade@fmu.ac.jp" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Fukushima Medical University, Fukushima, Japan" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Morfea generalizada pediátrica de aparición en el sitio de la inyección de la vacuna BCG" ] ] "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" => 1000 "Ancho" => 763 "Tamanyo" => 172041 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histological features included thickened collagen bundles in the dermis, fibrosis of septal tissues, and patchy lymphocytic infiltrates in the subcutaneous tissues.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">An 8-year-old girl was referred to our department with a linear sclerotic lesion on the upper left arm that had appeared several months earlier. The lesion had developed on the upper lateral aspect of the left arm, at the site of a Bacille Calmette-Guerin (BCG) vaccine, given more than 7 years earlier. It had gradually spread in a linear pattern and new sclerotic plaques appeared in increasing numbers over the subsequent months. Physical examination revealed a linear, slightly sclerotic plaque on the forehead (scleroderma en coup de sabre), linear pigmented, shiny plaques on the upper left arm, and sclerotic plaques on the right abdomen, left axilla, and back (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A–C). The girl also complained of muscle weakness in the upper left arm and occasional headaches. A biopsy specimen taken from the BCG vaccination site revealed thickened collagen bundles throughout the dermis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Patchy lymphocytic infiltrates and fibrosis of septal connective tissues were seen in the subcutaneous tissue. Laboratory tests revealed positive antinuclear antibodies (1:320, speckled and nucleolar) and values within the normal range for rheumatoid factor and anti-DNA, anti-centromere, and anti-U1RNP antibodies. Liver and kidney function, serum complement levels, creatine phosphokinase, aldolase, and myoglobin were also all within normal ranges. Three-dimensional computed tomography of the scalp did not reveal any abnormal findings. The patient was initially treated with oral prednisolone (15<span class="elsevierStyleHsp" style=""></span>mg/d), followed by the addition of methotrexate (6<span class="elsevierStyleHsp" style=""></span>mg/wk), which led to satisfactory results.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Morphea is sometimes triggered by local stimuli, such as minor trauma, irradiation, vaccination, implantation of silicon prostheses, needle biopsy, laparoscopy, and drug injections; it can also arise at the site of surgical and herpes zoster scars.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–7</span></a> The Koebner phenomenon is seen in various disorders, including morphea. The pathogenesis of this phenomenon has not yet been fully elucidated. Upon epidermal injury, several proinflammatory cytokines, such as interleukin 1, tumor necrosis factor α, and granulocyte macrophage-colony stimulating factor (GM-CSF), are released, possibly inducing further inflammation. Ueki<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> proposed a 2-step theory to explain the pathophysiology of the Koebner phenomenon. The first step, referred to as a nonspecific inflammatory step, would involve multiple environmentally induced factors such as cytokines, stress proteins, adhesion molecules, and autoantigens translocated from intracellular areas, while the second, disease-specific, step would involve disease-specific reactions mediated by T cells, B cells, autoantibodies, and immune complex deposition under the restriction of susceptible backgrounds.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Dermatological complications of BCG vaccination generally include induration and severe ulceration, but cases of granuloma annulare, abscesses, papular tuberculids, lupus vulgaris, and benign and malignant tumors have also been reported. Keloid formation at the BCG vaccination site is well known, but few cases of morphea have been reported to date.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a> In one of the cases, the lesions appeared on the shoulder a year after BCG vaccination, which was proposed as a possible trigger for the skin changes.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Another case was reported in a series of 7 cases of sclerodermatous conditions after bone marrow transplantation, but a detailed description was not provided.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Our patient developed linear scleroderma that started at the site of a BCG scar, without prior keloid scarring. Previous trauma may be associated with persistent inflammation and the release of various mediators such as cytokines and growth factors, or neurotransmitters from degenerative peripheral nerves.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 953 "Ancho" => 1000 "Tamanyo" => 119057 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Clinical appearance of the linear sclerotic plaque on the patient's arm. The Bacille Calmette-Guerin vaccination scar is shown by the arrow. (B) Linear sclerotic plaque on the forehead. (C) Morphea plaque on the right abdomen.</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" => 1000 "Ancho" => 763 "Tamanyo" => 172041 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Histological features included thickened collagen bundles in the dermis, fibrosis of septal tissues, and patchy lymphocytic infiltrates in the subcutaneous tissues.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Linear and multiple scleroatrophic condition of the lower limbs in a 7-month-old infant: etiopathogenic discussion" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "F. 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| 2022 Diciembre | 53 | 25 | 78 |
| 2022 Noviembre | 29 | 22 | 51 |
| 2022 Octubre | 32 | 29 | 61 |
| 2022 Septiembre | 35 | 33 | 68 |
| 2022 Agosto | 36 | 49 | 85 |
| 2022 Julio | 36 | 46 | 82 |
| 2022 Junio | 33 | 31 | 64 |
| 2022 Mayo | 34 | 38 | 72 |
| 2022 Abril | 48 | 31 | 79 |
| 2022 Marzo | 36 | 41 | 77 |
| 2022 Febrero | 47 | 32 | 79 |
| 2022 Enero | 67 | 53 | 120 |
| 2021 Diciembre | 43 | 31 | 74 |
| 2021 Noviembre | 64 | 43 | 107 |
| 2021 Octubre | 54 | 57 | 111 |
| 2021 Septiembre | 64 | 35 | 99 |
| 2021 Agosto | 86 | 23 | 109 |
| 2021 Julio | 81 | 29 | 110 |
| 2021 Junio | 30 | 30 | 60 |
| 2021 Mayo | 31 | 19 | 50 |
| 2021 Abril | 104 | 60 | 164 |
| 2021 Marzo | 49 | 19 | 68 |
| 2021 Febrero | 53 | 24 | 77 |
| 2021 Enero | 39 | 22 | 61 |
| 2020 Diciembre | 45 | 16 | 61 |
| 2020 Noviembre | 37 | 10 | 47 |
| 2020 Octubre | 22 | 11 | 33 |
| 2020 Septiembre | 36 | 6 | 42 |
| 2020 Agosto | 22 | 23 | 45 |
| 2020 Julio | 24 | 8 | 32 |
| 2020 Junio | 35 | 14 | 49 |
| 2020 Mayo | 28 | 19 | 47 |
| 2020 Abril | 36 | 20 | 56 |
| 2020 Marzo | 24 | 14 | 38 |
| 2020 Febrero | 4 | 0 | 4 |
| 2020 Enero | 1 | 4 | 5 |
| 2019 Diciembre | 4 | 5 | 9 |
| 2019 Noviembre | 0 | 1 | 1 |
| 2019 Octubre | 0 | 1 | 1 |
| 2019 Septiembre | 4 | 2 | 6 |
| 2019 Agosto | 0 | 1 | 1 |
| 2019 Julio | 0 | 4 | 4 |
| 2019 Junio | 2 | 2 | 4 |
| 2019 Mayo | 0 | 10 | 10 |
| 2019 Abril | 0 | 4 | 4 |
| 2019 Marzo | 2 | 4 | 6 |
| 2019 Febrero | 3 | 0 | 3 |
| 2019 Enero | 2 | 0 | 2 |
| 2018 Noviembre | 2 | 0 | 2 |
| 2018 Octubre | 2 | 0 | 2 |
| 2018 Septiembre | 5 | 0 | 5 |
| 2018 Marzo | 2 | 0 | 2 |
| 2018 Febrero | 29 | 3 | 32 |
| 2018 Enero | 27 | 5 | 32 |
| 2017 Diciembre | 30 | 5 | 35 |
| 2017 Noviembre | 19 | 2 | 21 |
| 2017 Octubre | 20 | 6 | 26 |
| 2017 Septiembre | 17 | 4 | 21 |
| 2017 Agosto | 15 | 8 | 23 |
| 2017 Julio | 20 | 4 | 24 |
| 2017 Junio | 28 | 8 | 36 |
| 2017 Mayo | 30 | 7 | 37 |
| 2017 Abril | 32 | 6 | 38 |
| 2017 Marzo | 12 | 24 | 36 |
| 2017 Febrero | 47 | 12 | 59 |
| 2017 Enero | 11 | 11 | 22 |
| 2016 Diciembre | 24 | 14 | 38 |
| 2016 Noviembre | 21 | 10 | 31 |
| 2016 Octubre | 15 | 11 | 26 |
| 2016 Septiembre | 0 | 6 | 6 |
| 2016 Agosto | 0 | 5 | 5 |
| 2016 Julio | 7 | 1 | 8 |
| 2016 Junio | 15 | 4 | 19 |
| 2016 Mayo | 20 | 1 | 21 |
| 2016 Abril | 11 | 18 | 29 |
| 2016 Marzo | 8 | 9 | 17 |
| 2016 Febrero | 7 | 14 | 21 |
| 2016 Enero | 12 | 10 | 22 |
| 2015 Diciembre | 10 | 6 | 16 |
| 2015 Noviembre | 14 | 3 | 17 |
| 2015 Octubre | 10 | 3 | 13 |
| 2015 Septiembre | 11 | 2 | 13 |
| 2015 Agosto | 0 | 1 | 1 |
| 2015 Julio | 2 | 8 | 10 |
| 2015 Junio | 2 | 2 | 4 |
| 2015 Mayo | 1 | 1 | 2 |
| 2015 Abril | 0 | 2 | 2 |
| 2015 Marzo | 0 | 3 | 3 |
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