Case and Research Letters
Eosinophilic Fasciitis: An Uncommon Cause of Edema
Fascitis eosinofílica: causa infrecuente de edemas
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Utrera-Busquets, J.C. Tardio, A. Curcio-Ruigomez, J. Borbujo-Martínez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M." "apellidos" => "Utrera-Busquets" "email" => array:1 [ 0 => "martautrerabusquets@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" => "J.C." "apellidos" => "Tardio" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Curcio-Ruigomez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Borbujo-Martínez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Cardiología, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Fascitis eosinofílica: causa infrecuente de edemas" ] ] "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" => 690 "Ancho" => 1034 "Tamanyo" => 73509 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Edema of the right arm; note the dimpled appearance of the skin of the inner arm and the increased diameter of the forearm.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The most common causes of generalized interstitial edema are heart failure, chronic kidney disease, and diseases of the liver. Edema, mainly affecting the extremities, is also the initial presenting symptom of scleroderma and other sclerodermiform syndromes. This type of edema is caused by fibrosis of the dermis and in its early stages can be suggestive of other pathologies.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We describe a patient with a history of mitral valve disease who, while awaiting valve surgery, was admitted for edema of the extremities of several months’ duration and was diagnosed with eosinophilic fasciitis (EF).</p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient was a 57-year-old man who presented with edema of 3 months’ duration that initially appeared on the lower limbs and subsequently developed on the hands and forearms. His past medical history included diabetes mellitus type 2 and hypercholesterolemia, and he had been diagnosed 6 years earlier with valvular heart disease (mitral valve prolapse) with severe mitral regurgitation. No abnormalities in left ventricular ejection fraction (LVEF) had been observed in cardiological examinations. The patient was admitted based on the suspicion that the edema had been triggered by a first episode of heart failure. He reported no dyspnea, orthopnea, or other related symptoms. Examination revealed a previously identified midsystolic murmur at the apex and hepatomegaly of 2<span class="elsevierStyleHsp" style=""></span>cm. An increase in the diameter of the legs and forearms was also observed, with induration and dimpling of the skin in these areas (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), as well as discrete erythema of the legs (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Blood tests showed eosinophilia, a slight elevation in the erythrocyte sedimentation rate (ESR), and hypergammaglobulinemia. All other parameters analyzed were normal. The previous diagnosis of mitral valve prolapse with severe mitral regurgitation was confirmed by transesophageal echocardiography. Left and right heart catheterization showed that both LVEF and pulmonary circuit pressure were normal. Given the contradictory clinical signs and the conflicting results of invasive cardiac tests, the patient was referred to the dermatology department. Given the suspicion of sclerodermiform syndrome a biopsy to the level of the muscular fascia was performed. The biopsy showed thickening and edema of the subcutaneous septa. The subcutaneous septa and superficial fascia contained a mild inflammatory infiltrate composed of lymphocytes, histiocytes, plasma cells, and eosinophils. The dermis and epidermis were intact (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Based on the diagnosis of EF the patient began prednisone treatment (60<span class="elsevierStyleHsp" style=""></span>mg/d), to which methotrexate was later added, resulting in clinical improvement and normalization of blood parameters. The patient is currently being treated with hydroxychloroquine and has a good quality of life, with no impairment of mobility and no change in valvular heart disease status.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">EF is a sclerodermiform syndrome that was first described by Shulman<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> in 1974. It is characterized by an initial phase of edema, erythema, and thickening of the skin, with symmetrical involvement of the extremities, although it can also affect the trunk and neck. After this initial phase the skin and subcutaneous tissue becomes indurated and the skin acquires an orange-peel-like appearance. EF can also present as morphea-like skin lesions; these 2 entities have been linked in various studies.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> It can also be associated with extracutaneous manifestations such as arthritis, joint stiffness, carpal tunnel syndrome, and muscle weakness. There have been isolated reports of systemic or visceral involvement. EF has been linked to blood disorders such as aplastic anemia, autoimmune anemia and thrombocytopenia, and lymphoproliferative diseases (lymphoma and multiple myeloma). It is accompanied by peripheral blood eosinophilia (up to 80%-90% of cases, depending on the series), increased ESR, and hypergammaglobulinemia, although the absence of any of these symptoms does not rule out the presence of the disease. While the etiology of EF is unknown, it has been associated with a history of intense exercise, trauma,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a><span class="elsevierStyleItalic">Borrelia burgdorferi</span> infection, and the use of drugs, such as simvastatin.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a> Confirmatory diagnosis is established by histological analysis of the skin, including the superficial muscle fascia. The following signs are typical: thickening of the septa of the subcutaneous tissue and of the fascia, which contain an inflammatory infiltrate of variable intensity that consists of lymphocytes, plasma cells, and occasionally eosinophils (the presence of which is not required for diagnosis) and is associated with dermal fibrosis. Recent studies have described the usefulness of magnetic resonance imaging in diagnosing and monitoring the progression of EF.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8,9</span></a> The differential diagnosis includes eosinophilia-myalgia syndrome, morphea, systemic scleroderma, and other sclerodermiform syndromes. As in the present case, the initial edema can be suggestive of other edema-causing conditions. Early treatment is essential to preserve mobility. The drugs of choice are oral corticosteroids, although hydroxychloroquine, methotrexate, and other immunosuppressive agents have also been used in isolated cases. Three predictors of treatment resistance have been identified: the presence of morphea-like skin lesions; early disease onset (patients under 12 years); and trunk involvement.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,10</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">We have reported a case of EF, a rare entity that presents diagnostic challenges and requires multidisciplinary management. Nondermatologist specialists should be familiar with the initial clinical features of this condition, including edema and erythema, in order to either rule out the disease or avoid delays in diagnosis, which can lead to further complications.</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: Utrera-Busquets M, Tardio J, Curcio-Ruigomez A, Borbujo-Martínez J. Fascitis eosinofílica: causa infrecuente de edemas. Actas Dermosifiliogr. 2014;105:626–628.</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" => 690 "Ancho" => 1034 "Tamanyo" => 73509 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Edema of the right arm; note the dimpled appearance of the skin of the inner arm and the increased diameter of the forearm.</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" => 737 "Ancho" => 982 "Tamanyo" => 84710 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Bilateral edema and erythema of both legs.</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" => 771 "Ancho" => 1033 "Tamanyo" => 310721 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histology showing thickened and edematous subcutaneous septa. Both the subcutaneous septa and the superficial fascia contain a mixed inflammatory infiltrate (hematoxylin-eosin, original magnification ×100).</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" => "An unusual case of generalized oedema" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S.N. Ingelbrecht" 1 => "H. Wynants" 2 => "G. Moorkens" 3 => "J.L. Gielen" 4 => "J.J. Martin" 5 => "W. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 11 | 13 | 24 |
| 2024 Octubre | 86 | 33 | 119 |
| 2024 Septiembre | 112 | 20 | 132 |
| 2024 Agosto | 111 | 47 | 158 |
| 2024 Julio | 114 | 42 | 156 |
| 2024 Junio | 141 | 43 | 184 |
| 2024 Mayo | 115 | 30 | 145 |
| 2024 Abril | 98 | 25 | 123 |
| 2024 Marzo | 106 | 29 | 135 |
| 2024 Febrero | 89 | 33 | 122 |
| 2024 Enero | 90 | 36 | 126 |
| 2023 Diciembre | 74 | 25 | 99 |
| 2023 Noviembre | 141 | 28 | 169 |
| 2023 Octubre | 107 | 22 | 129 |
| 2023 Septiembre | 115 | 32 | 147 |
| 2023 Agosto | 69 | 26 | 95 |
| 2023 Julio | 81 | 35 | 116 |
| 2023 Junio | 67 | 30 | 97 |
| 2023 Mayo | 73 | 28 | 101 |
| 2023 Abril | 87 | 23 | 110 |
| 2023 Marzo | 60 | 31 | 91 |
| 2023 Febrero | 59 | 24 | 83 |
| 2023 Enero | 55 | 31 | 86 |
| 2022 Diciembre | 74 | 41 | 115 |
| 2022 Noviembre | 31 | 30 | 61 |
| 2022 Octubre | 37 | 22 | 59 |
| 2022 Septiembre | 37 | 41 | 78 |
| 2022 Agosto | 27 | 38 | 65 |
| 2022 Julio | 31 | 37 | 68 |
| 2022 Junio | 26 | 25 | 51 |
| 2022 Mayo | 75 | 44 | 119 |
| 2022 Abril | 54 | 44 | 98 |
| 2022 Marzo | 52 | 55 | 107 |
| 2022 Febrero | 57 | 18 | 75 |
| 2022 Enero | 49 | 40 | 89 |
| 2021 Diciembre | 57 | 41 | 98 |
| 2021 Noviembre | 59 | 47 | 106 |
| 2021 Octubre | 77 | 45 | 122 |
| 2021 Septiembre | 68 | 41 | 109 |
| 2021 Agosto | 65 | 20 | 85 |
| 2021 Julio | 51 | 27 | 78 |
| 2021 Junio | 53 | 23 | 76 |
| 2021 Mayo | 52 | 25 | 77 |
| 2021 Abril | 82 | 48 | 130 |
| 2021 Marzo | 81 | 20 | 101 |
| 2021 Febrero | 59 | 23 | 82 |
| 2021 Enero | 31 | 8 | 39 |
| 2020 Diciembre | 32 | 7 | 39 |
| 2020 Noviembre | 21 | 12 | 33 |
| 2020 Octubre | 43 | 9 | 52 |
| 2020 Septiembre | 40 | 14 | 54 |
| 2020 Agosto | 19 | 18 | 37 |
| 2020 Julio | 21 | 11 | 32 |
| 2020 Junio | 32 | 22 | 54 |
| 2020 Mayo | 10 | 19 | 29 |
| 2020 Abril | 31 | 11 | 42 |
| 2020 Marzo | 19 | 17 | 36 |
| 2020 Febrero | 5 | 3 | 8 |
| 2020 Enero | 4 | 3 | 7 |
| 2019 Diciembre | 8 | 5 | 13 |
| 2019 Noviembre | 4 | 1 | 5 |
| 2019 Septiembre | 4 | 3 | 7 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 1 | 5 |
| 2019 Junio | 4 | 3 | 7 |
| 2019 Mayo | 6 | 15 | 21 |
| 2019 Abril | 2 | 0 | 2 |
| 2019 Marzo | 4 | 5 | 9 |
| 2019 Enero | 2 | 0 | 2 |
| 2018 Diciembre | 3 | 0 | 3 |
| 2018 Octubre | 2 | 0 | 2 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Febrero | 25 | 5 | 30 |
| 2018 Enero | 37 | 4 | 41 |
| 2017 Diciembre | 43 | 7 | 50 |
| 2017 Noviembre | 33 | 7 | 40 |
| 2017 Octubre | 20 | 9 | 29 |
| 2017 Septiembre | 32 | 10 | 42 |
| 2017 Agosto | 52 | 14 | 66 |
| 2017 Julio | 40 | 5 | 45 |
| 2017 Junio | 36 | 15 | 51 |
| 2017 Mayo | 24 | 11 | 35 |
| 2017 Abril | 23 | 11 | 34 |
| 2017 Marzo | 25 | 13 | 38 |
| 2017 Febrero | 14 | 4 | 18 |
| 2017 Enero | 17 | 1 | 18 |
| 2016 Diciembre | 32 | 10 | 42 |
| 2016 Noviembre | 38 | 10 | 48 |
| 2016 Octubre | 36 | 12 | 48 |
| 2016 Septiembre | 47 | 12 | 59 |
| 2016 Agosto | 39 | 13 | 52 |
| 2016 Julio | 24 | 3 | 27 |
| 2016 Junio | 9 | 3 | 12 |
| 2016 Mayo | 8 | 3 | 11 |
| 2016 Abril | 2 | 22 | 24 |
| 2016 Marzo | 5 | 1 | 6 |
| 2016 Febrero | 4 | 1 | 5 |
| 2016 Enero | 13 | 1 | 14 |
| 2015 Diciembre | 12 | 0 | 12 |
| 2015 Noviembre | 5 | 2 | 7 |
| 2015 Octubre | 11 | 2 | 13 |
| 2015 Septiembre | 16 | 4 | 20 |
| 2015 Agosto | 9 | 2 | 11 |
| 2015 Julio | 70 | 6 | 76 |
| 2015 Junio | 36 | 6 | 42 |
| 2015 Mayo | 46 | 8 | 54 |
| 2015 Abril | 19 | 9 | 28 |
| 2015 Marzo | 34 | 3 | 37 |
| 2015 Febrero | 21 | 4 | 25 |
| 2015 Enero | 13 | 5 | 18 |
| 2014 Diciembre | 14 | 5 | 19 |
| 2014 Noviembre | 9 | 5 | 14 |
| 2014 Octubre | 10 | 2 | 12 |
| 2014 Septiembre | 0 | 1 | 1 |
| 2014 Agosto | 2 | 2 | 4 |
| 2014 Julio | 2 | 2 | 4 |
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