Case and Research Letter
Bullous Sweet's syndrome with myositis
Síndrome de Sweet ampollar con miositis
K. Sato, T. Miura, M. Ohtsuka, T. Yamamoto
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Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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Sato, T. Miura, M. Ohtsuka, T. Yamamoto" "autores" => array:4 [ 0 => array:2 [ "nombre" => "K." "apellidos" => "Sato" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Miura" ] 2 => array:2 [ "nombre" => "M." "apellidos" => "Ohtsuka" ] 3 => 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" => "Síndrome de Sweet ampollar con miositis" ] ] "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" => 1218 "Ancho" => 1625 "Tamanyo" => 345842 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) Painful, erosive lesions on the upper chest with peripheral tense blisters (insert). (b) Histological features showing dense neutrophil infiltration throughout the edematous dermis. (c) Higher magnification showed neutrophil infiltration. (d) MRI examination revealed edematous swelling on the right gluteus maximus muscle.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 79-year-old female, who had suffered from myelodysplastic syndrome (MDS) (RAEB-1), was referred to our department complaining of painful lesions on the neck, chest and lower extremities with fever-up which appeared three weeks previously. She had received occasional transfusion for her MDS, but granulocyte-colony stimulating factor (G-CSF) was not administered. Physical examination showed ill-defined, tender edematous erythemas with bullous and erosive lesions on the chest (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). In addition, painful fresh-colored erythematous plaques were scattered on the lower extremities. Laboratory examination showed slightly elevated white blood cell counts (9100/μl with 27% Band, 29% Seg, 12% Lym, 4% Mono, 4% Eo, 9% Baso, and 10% Meta), and increased levels of erythrocyte sedimentation rate (136<span class="elsevierStyleHsp" style=""></span>mm/h) and C-reactive protein (9.0<span class="elsevierStyleHsp" style=""></span>mg/dl). A biopsy specimen from the chest showed dense neutrophilic infiltration throughout the edematous dermis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>c). Bacterial culture resulted sterile. Bone marrow biopsy revealed severe hypocellular bone marrow with a marked decrease of erythroblasts and megakaryocytes. After admission, systemic prednisolone (20<span class="elsevierStyleHsp" style=""></span>mg/day) was started which resulted in the improvement of skin lesions. However, during the course, she complained of severe muscle pain of the right thigh along with fever up to 39<span class="elsevierStyleHsp" style=""></span>°C. Examination by MRI showed edematous swelling on the right gluteus maximus muscle (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>d). Unfortunately, muscle biopsy was not performed, because her general conditions worsened. Serum creatine kinase level was not elevated and myositis was gradually improved without dose-up of prednisolone. However, her general condition was worsened, and she died of disseminated intravascular coagulation, renal failure, and complete A-V block one month after admission.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Sweet's syndrome is characterized by tender erythematous skin lesions accompanied by fever-up, in which inflammatory cells predominantly consisted of neutrophils infiltrate diffusely in the dermis. Sometimes Sweet's syndrome presents with atypical variants,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> and bullous variant is histologically characterized by extensive neutrophilic exocytosis and severe edema of the upper dermis. Whether cases of bullous Sweet's syndrome are commonly associated with hematological disorders is controversial.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">2,3</span></a> In the present case, bullous lesions were developed in a patient with active and severe MDS. The patient had no other apparent triggers such as upper airway or gastrointestinal infections and the use of new drugs, for the induction of Sweet's syndrome. Also, the patient developed non-bullous infiltrative erythema on the knee, and muscle tenderness during the course. So far, several cases of extracutaneous manifestations of Sweet's syndrome have been reported involving the lung, digestive tract, joints, lymph nodes, liver, spleen, eyes, central nervous system, and bone.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> Only several cases of neutrophilic myositis have been reported in association with neutrophilic dermatosis<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a>; however, to our knowledge, there have been no reports of neutrophilic myositis in association with bullous Sweet's syndrome. A previously reported case developed severe sterile neutrophilic myositis as the first manifestation of acute myelogenous leukemia.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> In the present case, myositis was developed soon after admission, which occurred almost concurrently with cutaneous manifestations. Because of the increased risk of infection, we did not escalate the dose of prednisolone; however, muscle lesions were transient and gradually improved. Because muscle biopsy was not carried out, it is uncertain that the patient developed neutrophilic myositis during the course. Hematoma was unlikely, but other factor such as infection was not denied, because drainage procedures were not performed. In such cases as immunocompromised patients like ours, it is often difficult to decide whether other organ involvement is caused by aseptic neutrophilic infiltration or infection.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0015" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interests.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1218 "Ancho" => 1625 "Tamanyo" => 345842 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) Painful, erosive lesions on the upper chest with peripheral tense blisters (insert). (b) Histological features showing dense neutrophil infiltration throughout the edematous dermis. (c) Higher magnification showed neutrophil infiltration. (d) MRI examination revealed edematous swelling on the right gluteus maximus muscle.</p>" ] ] ] "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" => "Bullous Sweet syndrome associated with an aseptic splenic abscess" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "S. Klinger" 1 => "N. Mathis" 2 => "S. 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| 2022 Noviembre | 13 | 25 | 38 |
| 2022 Octubre | 19 | 19 | 38 |
| 2022 Septiembre | 29 | 42 | 71 |
| 2022 Agosto | 22 | 29 | 51 |
| 2022 Julio | 23 | 43 | 66 |
| 2022 Junio | 27 | 27 | 54 |
| 2022 Mayo | 48 | 34 | 82 |
| 2022 Abril | 52 | 32 | 84 |
| 2022 Marzo | 61 | 37 | 98 |
| 2022 Febrero | 43 | 33 | 76 |
| 2022 Enero | 55 | 31 | 86 |
| 2021 Diciembre | 51 | 44 | 95 |
| 2021 Noviembre | 62 | 40 | 102 |
| 2021 Octubre | 73 | 48 | 121 |
| 2021 Septiembre | 54 | 43 | 97 |
| 2021 Agosto | 73 | 31 | 104 |
| 2021 Julio | 44 | 36 | 80 |
| 2021 Junio | 39 | 20 | 59 |
| 2021 Mayo | 58 | 39 | 97 |
| 2021 Abril | 76 | 41 | 117 |
| 2021 Marzo | 75 | 41 | 116 |
| 2021 Febrero | 58 | 32 | 90 |
| 2021 Enero | 40 | 19 | 59 |
| 2020 Diciembre | 26 | 21 | 47 |
| 2020 Noviembre | 42 | 22 | 64 |
| 2020 Octubre | 29 | 23 | 52 |
| 2020 Septiembre | 55 | 22 | 77 |
| 2020 Agosto | 38 | 24 | 62 |
| 2020 Julio | 38 | 19 | 57 |
| 2020 Junio | 34 | 43 | 77 |
| 2020 Mayo | 28 | 16 | 44 |
| 2020 Abril | 22 | 15 | 37 |
| 2020 Marzo | 22 | 12 | 34 |
| 2020 Febrero | 4 | 0 | 4 |
| 2020 Enero | 2 | 0 | 2 |
| 2019 Diciembre | 8 | 0 | 8 |
| 2019 Noviembre | 4 | 0 | 4 |
| 2019 Septiembre | 10 | 0 | 10 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 7 | 0 | 7 |
| 2019 Abril | 2 | 0 | 2 |
| 2019 Marzo | 2 | 0 | 2 |
| 2019 Febrero | 2 | 0 | 2 |
| 2018 Diciembre | 2 | 0 | 2 |
| 2018 Noviembre | 3 | 0 | 3 |
| 2018 Octubre | 2 | 0 | 2 |
| 2018 Septiembre | 2 | 0 | 2 |
| 2018 Abril | 1 | 0 | 1 |
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