Letter to the Editor
Unilateral lymphedema as first presentation of sarcoidosis
Linfedema unilateral como primera presentación de sarcoidosis
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"/1699258X/0000001200000001/v1_201601160037/S1699258X15000704/v1_201601160037/es/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Unilateral lymphedema as first presentation of sarcoidosis" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "59" "paginaFinal" => "60" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Senol Kobak, Ahmet Karaarslan, Hakan Aycan" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Senol" "apellidos" => "Kobak" "email" => array:1 [ 0 => "senolkobak@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" => "Ahmet" "apellidos" => "Karaarslan" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Hakan" "apellidos" => "Aycan" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Sifa University, Faculty of Medicine, Department of Rheumatology, Turkey" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Sifa University, Faculty of Medicine, Department of Orthopedics, Turkey" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfedema unilateral como primera presentación de sarcoidosis" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 962 "Ancho" => 950 "Tamanyo" => 94651 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Sacroiliac joints MRI showed bilateral sacroiliitis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sarcoidosis is a systemic disease characterized by the involvement of multiple tissues and organs with a non-calcified granuloma reaction, which is not yet well understood.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Although the exact pathogenesis of sarcoidosis is not known, it is currently accepted that, in genetically susceptible individuals, it is caused through alteration of the cellular immune response after exposure to an environmental, occupational or infectious agent.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> It is presented with bilateral hilar lymphadenopathies, infiltrations in the lung, skin and eye lesions. The pathognomonic histological finding is the presence of non-calcified granulomas.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> These granulomas may form in almost any organ in the body. Sarcoidosis is one of the most important causes of granulomatous lymphadenitis.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Development of lymphedema is the result of the involvement of lymph nodes and obstruction of lymphatic drainage. In this report we present a female case of sarcoidosis and ankylosing spondylitis co-occurrence that applied to our clinic with widespread lymphedema on her left leg. The patient is 50-year-old female who presented to our clinic with lymphedema and swelling started from the left inguinal region and spread to the whole left lower extremity. She had also complaints of inflammatory low back pain going on for the last 15 years. In her physical examination there was widespread lymphedema on her left foot, lymphadenopathies on her left inguinal region, restriction on her neck and hip range of motion and bilateral Fabere/Fadir test positivity. Laboratory studies showed erythrocyte sedimentation rate and C-reactive protein elevation, serum calcium and angiotensin converting enzyme (ACE) elevation and chronic disease anemia. There were widespread conglomerate lymphadenopathies in the left inguinal region according to the soft tissue ultrasonography examination. Thorax CT showed paratracheal, mediastinal and bilateral hilar lymphadenopathies (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Sacroiliac joint radiography showed bilateral joint space narrowing and sclerosis. Sacroiliac MRI scan was reported as chronic sacroiliitis with the presence of active lesions (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Genetic analysis showed that HLA-B27 was positive. Biopsy of the inguinal lymphadenopathies revealed granulomatous lymphadenitis and non-calcified granulomas. <span class="elsevierStyleItalic">M. tuberculosis</span> was not detected owing to her PPD was negative, acid-fast stain on tissue samples from the biopsy was negative. According to clinical, laboratory, histopathological and radiological findings, we diagnosed our patient as sarcoidosis and ankylosing spondylitis, then she received 40<span class="elsevierStyleHsp" style=""></span>mg of prednisolone per day. Lower extremity edema, inguinal lymph nodes and clinical complaints regressed during the follow up period. Control erythrocyte sedimentation rate and C-reactive protein were normal. Control thorax CT showed significant diametric regression of the hilar and bilateral mediastinal lymph nodes. Sarcoidosis may imitate different rheumatologic diseases and/or may be seen with them.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Sacroiliac joint involvement is a major joint involvement of sarcoidosis and it may be seen in 6–14% of the patients which causes the diagnostic confusion with ankylosing spondylitis.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6,7</span></a> HLA-B27 is helpful for the differential diagnosis besides the major method is the histopathological evaluation. Even if it is rare; like in our case, these two diseases may occur together. However this co-occurrence is not because of a common etiopathogenesis, but because of an incidental association. Granulomatous lymphadenitis is an important finding of sarcoidosis.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> The lymphadenopathies that develop in different regions cause organ and system dysfunctions. There are some reported sarcoidosis patients in the literature that were presented with lymphedema. Putkonen et al. has identified a female case that started with lower extremity lymphedema and they reported significant regression after treatment with corticosteroids.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Nathan et al. reported a 32-year-old black female case that suffered from foot lymphedema going on for the last 11 years.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> As in our case, the lymphedema development on these patients feet is because of the obstruction of the lymphatic drainage due to the involvement of the lymph nodes. Chronic, asymmetric feet edema presentation without venous obstruction should always suggest the probability of sarcoid lymphadenopathy for early diagnosis and treatment of this clinical condition. In conclusion, granulomatous lymphadenitis is an important clinical presentation of sarcoidosis. Different clinical findings may occur because of the compression of the conglomerated lymph nodes. For patients presented with compressive peripheral lymphadenopathy, sarcoidosis is a disease that should be kept in mind.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 778 "Ancho" => 995 "Tamanyo" => 127087 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Torax computed tomography showed bilateral hilar and mediastinal lymphadenopaties.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 962 "Ancho" => 950 "Tamanyo" => 94651 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Sacroiliac joints MRI showed bilateral sacroiliitis.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sarcoidosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "L.S. 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Elguezabel" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Arch Dermatol" "fecha" => "1974" "volumen" => "109" "paginaInicial" => "543" "paginaFinal" => "544" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/4819109" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/1699258X/0000001200000001/v1_201601160037/S1699258X15000613/v1_201601160037/en/main.assets" "Apartado" => array:4 [ "identificador" => "17506" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Cartas al editor" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/1699258X/0000001200000001/v1_201601160037/S1699258X15000613/v1_201601160037/en/main.pdf?idApp=UINPBA00004M&text.app=https://reumatologiaclinica.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1699258X15000613?idApp=UINPBA00004M" ]
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| 2024 Junio | 241 | 41 | 282 |
| 2024 Mayo | 200 | 38 | 238 |
| 2024 Abril | 151 | 26 | 177 |
| 2024 Marzo | 198 | 43 | 241 |
| 2024 Febrero | 136 | 21 | 157 |
| 2024 Enero | 172 | 29 | 201 |
| 2023 Diciembre | 161 | 34 | 195 |
| 2023 Noviembre | 163 | 30 | 193 |
| 2023 Octubre | 188 | 52 | 240 |
| 2023 Septiembre | 165 | 36 | 201 |
| 2023 Agosto | 190 | 29 | 219 |
| 2023 Julio | 151 | 31 | 182 |
| 2023 Junio | 147 | 27 | 174 |
| 2023 Mayo | 196 | 21 | 217 |
| 2023 Abril | 182 | 15 | 197 |
| 2023 Marzo | 274 | 42 | 316 |
| 2023 Febrero | 225 | 31 | 256 |
| 2023 Enero | 205 | 33 | 238 |
| 2022 Diciembre | 255 | 51 | 306 |
| 2022 Noviembre | 238 | 32 | 270 |
| 2022 Octubre | 207 | 43 | 250 |
| 2022 Septiembre | 219 | 56 | 275 |
| 2022 Agosto | 218 | 50 | 268 |
| 2022 Julio | 183 | 53 | 236 |
| 2022 Junio | 195 | 47 | 242 |
| 2022 Mayo | 259 | 58 | 317 |
| 2022 Abril | 174 | 68 | 242 |
| 2022 Marzo | 235 | 57 | 292 |
| 2022 Febrero | 224 | 47 | 271 |
| 2022 Enero | 198 | 40 | 238 |
| 2021 Diciembre | 153 | 37 | 190 |
| 2021 Noviembre | 148 | 56 | 204 |
| 2021 Octubre | 168 | 57 | 225 |
| 2021 Septiembre | 140 | 55 | 195 |
| 2021 Agosto | 180 | 59 | 239 |
| 2021 Julio | 150 | 42 | 192 |
| 2021 Junio | 206 | 58 | 264 |
| 2021 Mayo | 225 | 48 | 273 |
| 2021 Abril | 606 | 105 | 711 |
| 2021 Marzo | 292 | 55 | 347 |
| 2021 Febrero | 197 | 39 | 236 |
| 2021 Enero | 200 | 40 | 240 |
| 2020 Diciembre | 200 | 33 | 233 |
| 2020 Noviembre | 184 | 32 | 216 |
| 2020 Octubre | 135 | 29 | 164 |
| 2020 Septiembre | 190 | 36 | 226 |
| 2020 Agosto | 174 | 31 | 205 |
| 2020 Julio | 154 | 36 | 190 |
| 2020 Junio | 231 | 29 | 260 |
| 2020 Mayo | 178 | 33 | 211 |
| 2020 Abril | 141 | 24 | 165 |
| 2020 Marzo | 124 | 25 | 149 |
| 2020 Febrero | 141 | 41 | 182 |
| 2020 Enero | 147 | 39 | 186 |
| 2019 Diciembre | 111 | 34 | 145 |
| 2019 Noviembre | 133 | 21 | 154 |
| 2019 Octubre | 107 | 23 | 130 |
| 2019 Septiembre | 144 | 37 | 181 |
| 2019 Agosto | 127 | 25 | 152 |
| 2019 Julio | 99 | 17 | 116 |
| 2019 Junio | 112 | 44 | 156 |
| 2019 Mayo | 135 | 76 | 211 |
| 2019 Abril | 129 | 52 | 181 |
| 2019 Marzo | 135 | 33 | 168 |
| 2019 Febrero | 105 | 29 | 134 |
| 2019 Enero | 144 | 32 | 176 |
| 2018 Diciembre | 154 | 49 | 203 |
| 2018 Noviembre | 136 | 11 | 147 |
| 2018 Octubre | 123 | 13 | 136 |
| 2018 Septiembre | 127 | 10 | 137 |
| 2018 Agosto | 107 | 21 | 128 |
| 2018 Julio | 51 | 10 | 61 |
| 2018 Mayo | 5 | 1 | 6 |
| 2018 Abril | 79 | 12 | 91 |
| 2018 Marzo | 112 | 5 | 117 |
| 2018 Febrero | 78 | 11 | 89 |
| 2018 Enero | 56 | 11 | 67 |
| 2017 Diciembre | 74 | 12 | 86 |
| 2017 Noviembre | 78 | 12 | 90 |
| 2017 Octubre | 82 | 11 | 93 |
| 2017 Septiembre | 60 | 10 | 70 |
| 2017 Agosto | 66 | 11 | 77 |
| 2017 Julio | 52 | 15 | 67 |
| 2017 Junio | 65 | 19 | 84 |
| 2017 Mayo | 91 | 15 | 106 |
| 2017 Abril | 57 | 37 | 94 |
| 2017 Marzo | 63 | 23 | 86 |
| 2017 Febrero | 109 | 11 | 120 |
| 2017 Enero | 33 | 7 | 40 |
| 2016 Diciembre | 81 | 22 | 103 |
| 2016 Noviembre | 75 | 9 | 84 |
| 2016 Octubre | 119 | 19 | 138 |
| 2016 Septiembre | 135 | 19 | 154 |
| 2016 Agosto | 29 | 9 | 38 |
| 2016 Julio | 23 | 18 | 41 |
| 2016 Mayo | 1 | 0 | 1 |
| 2016 Marzo | 1 | 0 | 1 |
| 2016 Febrero | 1 | 0 | 1 |
| 2016 Enero | 3 | 0 | 3 |
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