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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 10 | 12 | 22 |
| 2024 October | 86 | 33 | 119 |
| 2024 September | 112 | 20 | 132 |
| 2024 August | 111 | 47 | 158 |
| 2024 July | 114 | 42 | 156 |
| 2024 June | 141 | 43 | 184 |
| 2024 May | 115 | 30 | 145 |
| 2024 April | 98 | 25 | 123 |
| 2024 March | 106 | 29 | 135 |
| 2024 February | 89 | 33 | 122 |
| 2024 January | 90 | 36 | 126 |
| 2023 December | 74 | 25 | 99 |
| 2023 November | 141 | 28 | 169 |
| 2023 October | 107 | 22 | 129 |
| 2023 September | 115 | 32 | 147 |
| 2023 August | 69 | 26 | 95 |
| 2023 July | 81 | 35 | 116 |
| 2023 June | 67 | 30 | 97 |
| 2023 May | 73 | 28 | 101 |
| 2023 April | 87 | 23 | 110 |
| 2023 March | 60 | 31 | 91 |
| 2023 February | 59 | 24 | 83 |
| 2023 January | 55 | 31 | 86 |
| 2022 December | 74 | 41 | 115 |
| 2022 November | 31 | 30 | 61 |
| 2022 October | 37 | 22 | 59 |
| 2022 September | 37 | 41 | 78 |
| 2022 August | 27 | 38 | 65 |
| 2022 July | 31 | 37 | 68 |
| 2022 June | 26 | 25 | 51 |
| 2022 May | 75 | 44 | 119 |
| 2022 April | 54 | 44 | 98 |
| 2022 March | 52 | 55 | 107 |
| 2022 February | 57 | 18 | 75 |
| 2022 January | 49 | 40 | 89 |
| 2021 December | 57 | 41 | 98 |
| 2021 November | 59 | 47 | 106 |
| 2021 October | 77 | 45 | 122 |
| 2021 September | 68 | 41 | 109 |
| 2021 August | 65 | 20 | 85 |
| 2021 July | 51 | 27 | 78 |
| 2021 June | 53 | 23 | 76 |
| 2021 May | 52 | 25 | 77 |
| 2021 April | 82 | 48 | 130 |
| 2021 March | 81 | 20 | 101 |
| 2021 February | 59 | 23 | 82 |
| 2021 January | 31 | 8 | 39 |
| 2020 December | 32 | 7 | 39 |
| 2020 November | 21 | 12 | 33 |
| 2020 October | 43 | 9 | 52 |
| 2020 September | 40 | 14 | 54 |
| 2020 August | 19 | 18 | 37 |
| 2020 July | 21 | 11 | 32 |
| 2020 June | 32 | 22 | 54 |
| 2020 May | 10 | 19 | 29 |
| 2020 April | 31 | 11 | 42 |
| 2020 March | 19 | 17 | 36 |
| 2020 February | 5 | 3 | 8 |
| 2020 January | 4 | 3 | 7 |
| 2019 December | 8 | 5 | 13 |
| 2019 November | 4 | 1 | 5 |
| 2019 September | 4 | 3 | 7 |
| 2019 August | 4 | 0 | 4 |
| 2019 July | 4 | 1 | 5 |
| 2019 June | 4 | 3 | 7 |
| 2019 May | 6 | 15 | 21 |
| 2019 April | 2 | 0 | 2 |
| 2019 March | 4 | 5 | 9 |
| 2019 January | 2 | 0 | 2 |
| 2018 December | 3 | 0 | 3 |
| 2018 October | 2 | 0 | 2 |
| 2018 September | 4 | 0 | 4 |
| 2018 February | 25 | 5 | 30 |
| 2018 January | 37 | 4 | 41 |
| 2017 December | 43 | 7 | 50 |
| 2017 November | 33 | 7 | 40 |
| 2017 October | 20 | 9 | 29 |
| 2017 September | 32 | 10 | 42 |
| 2017 August | 52 | 14 | 66 |
| 2017 July | 40 | 5 | 45 |
| 2017 June | 36 | 15 | 51 |
| 2017 May | 24 | 11 | 35 |
| 2017 April | 23 | 11 | 34 |
| 2017 March | 25 | 13 | 38 |
| 2017 February | 14 | 4 | 18 |
| 2017 January | 17 | 1 | 18 |
| 2016 December | 32 | 10 | 42 |
| 2016 November | 38 | 10 | 48 |
| 2016 October | 36 | 12 | 48 |
| 2016 September | 47 | 12 | 59 |
| 2016 August | 39 | 13 | 52 |
| 2016 July | 24 | 3 | 27 |
| 2016 June | 9 | 3 | 12 |
| 2016 May | 8 | 3 | 11 |
| 2016 April | 2 | 22 | 24 |
| 2016 March | 5 | 1 | 6 |
| 2016 February | 4 | 1 | 5 |
| 2016 January | 13 | 1 | 14 |
| 2015 December | 12 | 0 | 12 |
| 2015 November | 5 | 2 | 7 |
| 2015 October | 11 | 2 | 13 |
| 2015 September | 16 | 4 | 20 |
| 2015 August | 9 | 2 | 11 |
| 2015 July | 70 | 6 | 76 |
| 2015 June | 36 | 6 | 42 |
| 2015 May | 46 | 8 | 54 |
| 2015 April | 19 | 9 | 28 |
| 2015 March | 34 | 3 | 37 |
| 2015 February | 21 | 4 | 25 |
| 2015 January | 13 | 5 | 18 |
| 2014 December | 14 | 5 | 19 |
| 2014 November | 9 | 5 | 14 |
| 2014 October | 10 | 2 | 12 |
| 2014 September | 0 | 1 | 1 |
| 2014 August | 2 | 2 | 4 |
| 2014 July | 2 | 2 | 4 |
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