Scientific Letter
Pleural Effusion: A Rare Manifestation of Sarcoidosis
Derrame pleural: una presentación poco frecuente de sarcoidosis
María Fontecha Ortega
, Sergio Julio Rodríguez Álvarez, Jose Luis García Satué
Corresponding author
Servicio de Medicina Interna, Hospital Universitario Getafe, Getafe, Madrid, Spain
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B) Radiografía de tórax lateral, se observan infiltrados alveolares parcheados en LM y LSI. Derrame pleural bilateral de predominio derecho. Adenopatías hiliares y mediastínicas en rango patológico. C) Imagen axial de la TC torácica con CIV, se observa el componente adenopático en compartimentos mediastínicos, hiliares bilaterales y periesofágico. 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(B) Radiograph of lateral thorax, showing patchy alveolar infiltrates in the left middle and upper lobes. Predominantly right-sided bilateral pleural effusion. Hilar and mediastinal lymphadenopathy of pathological appearance. (C) Axial image of chest CT with intravenous contrast medium showing lymphadenopathy component in the mediastinal, bilateral hilar and periesophageal compartments. Bilateral alveolar involvement predominantly in the subpleural region of both bases, with accumulated nodal lesions and patchy air bronchogram in the subpleural region.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sarcoidosis is a granulomatous disease of unknown origin. It most commonly affects the lungs, while associated extrapulmonary involvement can be variable. However, pleural involvement, with or without sarcoid pleural effusion, is rare.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> Moreover, non-specific systemic symptoms that confound the initial clinical suspicion even further are not uncommon.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Diagnosis of suspected disease is based on clinical and radiological findings and histological confirmation of non-caseifying epithelioid granulomas. Treatment is still currently based on corticosteroids, which are prescribed on the basis of clinical compromise, progression and/or relapses.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Disease progression and prognosis are variable.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> As the incidence of this entity is low, we report 1 case of sarcoidosis with PE, and discuss the importance of a differential diagnosis with tuberculous infection.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 38-year-old man, native of Ghana, diagnosed in 2003 with latent tuberculosis (TB), no chemoprophylaxis administered. Episodes of malaria during infancy. He consulted due to a 2-month history of cough, with bloody expectoration, dyspnea, pleuritic chest pain, with weight loss, Afebrile. Of note on physical examination were hypophonesis in the right lung base with crackles and rhonchi up to the middle fields in both sides. Chest radiography revealed bilateral multiple basal alveolar infiltrates with hila of nodular appearance, suggestive of lymphadenopathies, along with right PE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A and B). Of note on clinical laboratory tests: LDH 134, with reduced proteins and albumin, CRP 63<span class="elsevierStyleHsp" style=""></span>mg/dl and ESR 47. Protein electrophoresis spectrum with inflammatory pattern with no monoclonal peak, negative autoimmunity and negative HIV and HCV serologies, previous HBV and HAV. ACE 89.2 (normal<50). Negative for Mantoux booster phenomenon. Serous fluid obtained on thoracocentesis consistent with predominantly lymphocytic exudate, ADA 72. Mycobacteria and fungus-specific staining, cultures and molecular techniques were negative; immunophenotyping showed no clonality but did reveal a significant T cell population with a CD4+/CD8+ ratio of 4/1 (>3.5). After a chest-abdominal PET-CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C), a bronchoscopy was performed and a transbronchial biopsy obtained, showing necrotizing non-caseifying epithelioid granulomas. Final diagnosis of pulmonary sarcoidosis stage <span class="elsevierStyleSmallCaps">II</span> with secondary PE.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">The prevalence of PE in published series of sarcoidosis patients is rare, at between 1% and 2%,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> although some groups have reported rates of up to 8%–10%.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> Pleural involvement can appear at any age, but occurs mostly between the ages of 41 and 60 years, with no difference between sexes. It is usually mild-moderate, generally right-sided (48.3%), and occurs in disease stages <span class="elsevierStyleSmallCaps">I</span> and <span class="elsevierStyleSmallCaps">II</span>. It characteristically presents as serous exudate, although 2 distinct types have been described, depending on the etiological pathological mechanism. Lymphocytes are predominant (typically with a CD4+/CD8+ ratio between 2.61 and 8.6).<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Cases described in the literature<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> do not state whether patients previously presented a positive PPD, as was the case with our patient, which became negative during the study of the clinical symptoms of sarcoidosis. This effect, known as “immunological paradox”,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> consists of an immune-mediated phenomenon in which, despite intense local inflammation, anergy can develop as a result of dysregulation of the T cells involved in the immunopathology of the disease.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In view of our patient's previous positive PPD and extremely high ADA in PE, active tuberculous infection had to be reliably ruled out, so we performed mycobacteria IGRA, PCR, stains and cultures for 8 weeks in pleural fluid, that were all negative. Sputum smear in bronchial aspirate and urine, and closed pleural biopsy with mycobacterial PCR, cultures and stains were also negative. In view of the high initial suspicion of TB due to the history of untreated latent TB, tuberculostatic treatment was attempted for 10 days while awaiting results, but response was unsatisfactory. The microbiological results, when obtained, were negative, so we strongly suspected pleuropulmonary sarcoidosis in a black patient and began treatment with corticosteroids. Clinical symptoms and laboratory inflammatory parameters improved, and PE resolved, confirming the initially suspected diagnosis.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Finally, given the evidence of previous exposure to TB and current corticosteroid therapy, our patient was given chemoprophylaxis on the assumption that the PPD skin test was a false negative associated with immune system changes due to the disease itself.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Discussion" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Fontecha Ortega M, Rodríguez Álvarez SJ, García Satué JL. Derrame pleural: una presentación poco frecuente de sarcoidosis. Arch Bronconeumol. 2017;53:170–171.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1524 "Ancho" => 1350 "Tamanyo" => 273612 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) PA chest radiograph. (B) Radiograph of lateral thorax, showing patchy alveolar infiltrates in the left middle and upper lobes. Predominantly right-sided bilateral pleural effusion. Hilar and mediastinal lymphadenopathy of pathological appearance. (C) Axial image of chest CT with intravenous contrast medium showing lymphadenopathy component in the mediastinal, bilateral hilar and periesophageal compartments. Bilateral alveolar involvement predominantly in the subpleural region of both bases, with accumulated nodal lesions and patchy air bronchogram in the subpleural region.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sarcoidosis pulmonar" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A. Mussetti" 1 => "L. Vignoli" 2 => "P. Curbelo" 3 => "E. Meerovich" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Neumol Cir Torax" "fecha" => "2006" "volumen" => "65" "numero" => "Suppl. 3" "paginaInicial" => "S36" "paginaFinal" => "S46" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Derrame pleural sarcoideo" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "N. Rodríguez" 1 => "C. Rábade" 2 => "L. 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Teirstein" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1056/NEJMra071714" "Revista" => array:6 [ "tituloSerie" => "N Engl J Med" "fecha" => "2007" "volumen" => "357" "paginaInicial" => "2153" "paginaFinal" => "2165" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18032765" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pleural effusion in a series of 181 outpatients with sarcoidosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "J.T. Huggins" 1 => "P. Doelken" 2 => "S.A. Sahn" 3 => "L. King" 4 => "M.A. Judson" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1378/chest.129.6.1599" "Revista" => array:6 [ "tituloSerie" => "Chest" "fecha" => "2006" "volumen" => "129" "paginaInicial" => "1599" "paginaFinal" => "1604" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/16778281" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Derrame pleural y sarcoidosis: una asociación poco frecuente" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "L. Ferreiro" 1 => "E. San José" 2 => "F.J. González-Barcala" 3 => "A. Suárez-Antelo" 4 => "M.E. Toubes" 5 => "L. Valdés" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.arbres.2013.07.020" "Revista" => array:6 [ "tituloSerie" => "Arch Bronconeumol" "fecha" => "2014" "volumen" => "50" "paginaInicial" => "554" "paginaFinal" => "556" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/24565689" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005300000003/v1_201703240112/S1579212916303445/v1_201703240112/en/main.assets" "Apartado" => array:4 [ "identificador" => "49861" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005300000003/v1_201703240112/S1579212916303445/v1_201703240112/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212916303445?idApp=UINPBA00003Z" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 4 | 4 | 8 |
| 2024 October | 42 | 22 | 64 |
| 2024 September | 31 | 14 | 45 |
| 2024 August | 73 | 35 | 108 |
| 2024 July | 42 | 26 | 68 |
| 2024 June | 69 | 28 | 97 |
| 2024 May | 84 | 31 | 115 |
| 2024 April | 45 | 32 | 77 |
| 2024 March | 45 | 20 | 65 |
| 2024 February | 32 | 21 | 53 |
| 2023 March | 7 | 3 | 10 |
| 2023 February | 52 | 19 | 71 |
| 2023 January | 46 | 33 | 79 |
| 2022 December | 55 | 29 | 84 |
| 2022 November | 46 | 25 | 71 |
| 2022 October | 47 | 42 | 89 |
| 2022 September | 58 | 29 | 87 |
| 2022 August | 52 | 37 | 89 |
| 2022 July | 48 | 39 | 87 |
| 2022 June | 67 | 40 | 107 |
| 2022 May | 66 | 35 | 101 |
| 2022 April | 114 | 35 | 149 |
| 2022 March | 96 | 40 | 136 |
| 2022 February | 71 | 31 | 102 |
| 2022 January | 85 | 40 | 125 |
| 2021 December | 65 | 53 | 118 |
| 2021 November | 81 | 54 | 135 |
| 2021 October | 71 | 65 | 136 |
| 2021 September | 49 | 83 | 132 |
| 2021 August | 42 | 35 | 77 |
| 2021 July | 37 | 36 | 73 |
| 2021 June | 46 | 40 | 86 |
| 2021 May | 77 | 42 | 119 |
| 2021 April | 141 | 99 | 240 |
| 2021 March | 41 | 24 | 65 |
| 2021 February | 37 | 31 | 68 |
| 2021 January | 33 | 24 | 57 |
| 2020 December | 22 | 30 | 52 |
| 2020 November | 43 | 32 | 75 |
| 2020 October | 35 | 20 | 55 |
| 2020 September | 28 | 14 | 42 |
| 2020 August | 31 | 13 | 44 |
| 2020 July | 54 | 33 | 87 |
| 2020 June | 25 | 8 | 33 |
| 2020 May | 44 | 16 | 60 |
| 2020 April | 21 | 25 | 46 |
| 2020 March | 27 | 11 | 38 |
| 2020 February | 43 | 21 | 64 |
| 2020 January | 28 | 23 | 51 |
| 2019 December | 39 | 20 | 59 |
| 2019 November | 25 | 21 | 46 |
| 2019 October | 27 | 15 | 42 |
| 2019 September | 36 | 22 | 58 |
| 2019 August | 44 | 16 | 60 |
| 2019 July | 31 | 15 | 46 |
| 2019 June | 30 | 26 | 56 |
| 2019 May | 85 | 26 | 111 |
| 2019 April | 38 | 47 | 85 |
| 2019 March | 38 | 33 | 71 |
| 2019 February | 22 | 25 | 47 |
| 2019 January | 26 | 18 | 44 |
| 2018 December | 26 | 14 | 40 |
| 2018 November | 101 | 21 | 122 |
| 2018 October | 89 | 25 | 114 |
| 2018 September | 14 | 7 | 21 |
| 2018 May | 25 | 1 | 26 |
| 2018 April | 23 | 7 | 30 |
| 2018 March | 15 | 6 | 21 |
| 2018 February | 18 | 14 | 32 |
| 2018 January | 23 | 17 | 40 |
| 2017 December | 23 | 7 | 30 |
| 2017 November | 25 | 16 | 41 |
| 2017 October | 26 | 16 | 42 |
| 2017 September | 18 | 8 | 26 |
| 2017 August | 27 | 14 | 41 |
| 2017 July | 28 | 15 | 43 |
| 2017 June | 41 | 23 | 64 |
| 2017 May | 1 | 1 | 2 |
| 2017 April | 2 | 0 | 2 |
| 2017 January | 0 | 1 | 1 |
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