Letter to the Editor
Unilateral lymphedema as first presentation of sarcoidosis
Linfedema unilateral como primera presentación de sarcoidosis
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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" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 778 "Ancho" => 995 "Tamanyo" => 121825 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Torax computed tomography showed bilateral hilar and mediastinal lymphadenopaties.</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" => 121825 ] ] "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" => 89596 ] ] "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" => "/21735743/0000001200000001/v1_201601240019/S2173574316000101/v1_201601240019/en/main.assets" "Apartado" => array:4 [ "identificador" => "8400" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735743/0000001200000001/v1_201601240019/S2173574316000101/v1_201601240019/en/main.pdf?idApp=UINPBA00004M&text.app=https://reumatologiaclinica.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574316000101?idApp=UINPBA00004M" ]
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| 2024 August | 71 | 28 | 99 |
| 2024 July | 66 | 35 | 101 |
| 2024 June | 78 | 34 | 112 |
| 2024 May | 60 | 34 | 94 |
| 2024 April | 60 | 29 | 89 |
| 2024 March | 66 | 24 | 90 |
| 2024 February | 64 | 29 | 93 |
| 2024 January | 47 | 28 | 75 |
| 2023 December | 51 | 41 | 92 |
| 2023 November | 71 | 46 | 117 |
| 2023 October | 43 | 45 | 88 |
| 2023 September | 76 | 46 | 122 |
| 2023 August | 65 | 15 | 80 |
| 2023 July | 50 | 30 | 80 |
| 2023 June | 51 | 24 | 75 |
| 2023 May | 62 | 25 | 87 |
| 2023 April | 43 | 6 | 49 |
| 2023 March | 61 | 22 | 83 |
| 2023 February | 47 | 30 | 77 |
| 2023 January | 56 | 28 | 84 |
| 2022 December | 63 | 26 | 89 |
| 2022 November | 57 | 39 | 96 |
| 2022 October | 68 | 31 | 99 |
| 2022 September | 88 | 36 | 124 |
| 2022 August | 34 | 37 | 71 |
| 2022 July | 43 | 35 | 78 |
| 2022 June | 32 | 35 | 67 |
| 2022 May | 56 | 36 | 92 |
| 2022 April | 56 | 37 | 93 |
| 2022 March | 62 | 48 | 110 |
| 2022 February | 57 | 19 | 76 |
| 2022 January | 40 | 32 | 72 |
| 2021 December | 33 | 39 | 72 |
| 2021 November | 40 | 45 | 85 |
| 2021 October | 32 | 52 | 84 |
| 2021 September | 38 | 31 | 69 |
| 2021 August | 34 | 35 | 69 |
| 2021 July | 16 | 27 | 43 |
| 2021 June | 30 | 36 | 66 |
| 2021 May | 59 | 54 | 113 |
| 2021 April | 104 | 46 | 150 |
| 2021 March | 69 | 14 | 83 |
| 2021 February | 33 | 17 | 50 |
| 2021 January | 31 | 20 | 51 |
| 2020 December | 32 | 23 | 55 |
| 2020 November | 41 | 17 | 58 |
| 2020 October | 39 | 12 | 51 |
| 2020 September | 21 | 19 | 40 |
| 2020 August | 17 | 16 | 33 |
| 2020 July | 23 | 10 | 33 |
| 2020 June | 20 | 15 | 35 |
| 2020 May | 32 | 9 | 41 |
| 2020 April | 17 | 14 | 31 |
| 2020 March | 9 | 5 | 14 |
| 2018 December | 2 | 0 | 2 |
| 2018 May | 1 | 1 | 2 |
| 2018 April | 19 | 8 | 27 |
| 2018 March | 21 | 5 | 26 |
| 2018 February | 16 | 4 | 20 |
| 2018 January | 13 | 3 | 16 |
| 2017 December | 18 | 2 | 20 |
| 2017 November | 14 | 4 | 18 |
| 2017 October | 9 | 3 | 12 |
| 2017 September | 13 | 13 | 26 |
| 2017 August | 12 | 7 | 19 |
| 2017 July | 16 | 14 | 30 |
| 2017 June | 23 | 8 | 31 |
| 2017 May | 31 | 10 | 41 |
| 2017 April | 29 | 7 | 36 |
| 2017 March | 26 | 13 | 39 |
| 2017 February | 14 | 12 | 26 |
| 2017 January | 16 | 4 | 20 |
| 2016 December | 39 | 12 | 51 |
| 2016 November | 27 | 3 | 30 |
| 2016 October | 26 | 15 | 41 |
| 2016 September | 30 | 7 | 37 |
| 2016 August | 27 | 6 | 33 |
| 2016 July | 15 | 9 | 24 |
| 2016 June | 1 | 0 | 1 |
| 2016 April | 1 | 0 | 1 |
| 2016 February | 1 | 0 | 1 |
| 2016 January | 2 | 0 | 2 |
Show all
