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Pleuroparenchymal fibroelastosis: A new entity of interstitial pneumonia related to connective tissue diseases
Fibroelastosis pleuroparenquimatosa: un nuevo tipo de neumonía instersticial asociada a conectivopatías
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"patricia.moran@salud.madrid.org" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Javier" "apellidos" => "Bachiller-Corral" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Luis" "apellidos" => "Gorospe Sarasúa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Carlos" "apellidos" => "de la Puente Bujidos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital Universitario Ramón y Cajal, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Radiodiagnóstico, Hospital Universitario Ramón y Cajal, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Fibroelastosis pleuroparenquimatosa: un nuevo tipo de neumonía instersticial asociada a conectivopatías" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 617 "Ancho" => 1500 "Tamanyo" => 121997 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Transversal pulmonary CTA slice. (B) Coronal CTA pulmonary slice. In both slices the presence of subpleural consolidations may be seen, together with predominantly apical bilateral pleural thickening.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pleuroparenchymal fibroelastosis (PPFE) is a new entity that was recently included in the group of rare or infrequent idiopathic interstitial pneumonias.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is characterised by the development of an elastic fibre-rich fibrosis that affects the pleura and subpleural pulmonary parenchyma, predominantly in the upper lobes.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It may be idiopathic or secondary to multiple processes, including systemic autoimmune diseases.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 52 year old women from Peru who has lived in Spain for 23 years. She works as a home help and has no relevant family or personal history. A year ago arthralgia when moving commenced together with xerostomy but without xerophthalmia or any other associated symptom. Physical examination, cardiopulmonary auscultation and musculoskeletal and cutaneous examination were normal. Haemogram, general biochemistry and creatine kinase (111 U/l) were normal. IgG immunoglobulin in serum was raised (2.410 mg/dl). FR was positive (242 UI/mL), ACPA was negative, ANA was positive (1/640), anti-Ro was positive (240 UI/mL), anti-La was positive (75 UI/mL) and anti-DNA was negative. The following complementary tests were requested: serological tests for VHC and VHB, Interferon Gamma Release Assay (IGRA), sacroiliac X-ray imaging, echocardiogram, specific myopathy antibodies (anti-MI2, anti-SRP, anti-PM-SCL, anti-PL7, anti-PL12, anti-KU, anti-OJ and anti-EJ) and capillaroscopy, all of which were normal or negative.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Salivary gland biopsy revealed the presence of multiple areas of lymphocyte infiltration. She was diagnosed primary Sjögren syndrome on the basis of ACR-EULAR 2017 criteria.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Thoracic X-ray imaging showed pleural thickening and fibrosis in the apical cap, and pulmonary TCA imaging showed subpleural consolidations together with predominantly bilateral apical pleural thickening, leading to a moderate loss of volume in both upper lobes, all of which is compatible with PPFE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Respiratory function tests including CO diffusion were normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Treatment commenced with 5 mg/day prednisone, in spite of which the patient described worsening of the xerostomy and a subjective sensation of dyspnoea during moderate efforts. A second pulmonary CTA showed no radiological progression after a 6 month follow-up.</p><p id="par0025" class="elsevierStylePara elsevierViewall">PPFE was first described in 1992 by Amitani et al. as idiopathic pulmonary fibrosis of the upper fields, and in 2004 it was recognised by Frankel et al. as a new clinical-pathological entity. Since then approximately 120 cases have been described worldwide, above all in Asiatic populations.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Of all these cases, about 20% were associated with connective tissue pathologies.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a><span class="elsevierStyleBold">I</span>n 2013 this entity was included in the classification of idiopathic interstitial pneumonias of the American Thoracic Society/European Respiratory Society, within the category of rare interstitial pneumonias.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">This disease occurs in adults with an average age of around 57 years, affecting both sexes equally<span class="elsevierStyleSup">1</span> and without showing any association with smoking.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Its symptoms are usually dyspnoea, unproductive cough, pleuritic pain or weight loss.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> This entity may be idiopathic or secondary to multiple processes: infections, radiation, transplant or neoplasias, including rheumatological diseases<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> such as rheumatoid arthritis, myopathies, scleroderma or Sjögren's syndrome.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The characteristic findings in CTA imaging are the bilateral presence of irregular pleural thickening and fibrotic changes in upper fields of the subpleural parenchyma, and it may coexist with other radiological patterns of pulmonary interstitial disease in different areas of the parenchyma. Anatomic-pathological findings are the presence of pleural and intra-alveolar fibrosis together with elastosis of the alveolar septa.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Patients with PPFE are at higher risk of pneumomediastinum or pneumothorax,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> either spontaneously or after a pulmonary biopsy for definitive diagnosis based on anatomopathological findings.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> This is why the utility of cryobiopsy as a less invasive diagnostic technique is being evaluated, as it reduces complications of this type.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Differential diagnosis must be considered against other entities which are found radiologically to predominantly affect the upper lobes, including sarcoidosis or tuberculosis.</p><p id="par0045" class="elsevierStylePara elsevierViewall">PPFE has a highly variable prognosis, and its evolution is sometimes extremely severe.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Although a case has been described that improved with pirfenidone,<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> in general there is no effective treatment. The main causes of death are the progression of the disease and respiratory infections.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Three cases have been published to date in Spain, all of which evolved unfavourably and required lung transplant.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,9</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">To conclude, PPFE is a new entity that has recently been included among the interstitial lung diseases, and it may be associated with systemic autoimmune diseases. This is why it is important to be aware of its clinical and radiological characteristics, suspecting it in patients with interstitial pulmonary involvement that is predominantly in the upper fields. It is also highly important to use a multidisciplinary approach to improve its detection and treatment.</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: Morán Álvarez P, Bachiller-Corral J, Gorospe Sarasúa L, de la Puente Bujidos C. Fibroelastosis pleuroparenquimatosa: un nuevo tipo de neumonía instersticial asociada a conectivopatías. Reumatol Clin. 2020;16:513–514.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 617 "Ancho" => 1500 "Tamanyo" => 121997 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Transversal pulmonary CTA slice. (B) Coronal CTA pulmonary slice. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 4 | 4 | 8 |
| 2024 October | 52 | 26 | 78 |
| 2024 September | 49 | 33 | 82 |
| 2024 August | 75 | 53 | 128 |
| 2024 July | 57 | 45 | 102 |
| 2024 June | 60 | 35 | 95 |
| 2024 May | 73 | 33 | 106 |
| 2024 April | 55 | 35 | 90 |
| 2024 March | 58 | 30 | 88 |
| 2024 February | 107 | 28 | 135 |
| 2024 January | 58 | 25 | 83 |
| 2023 December | 35 | 22 | 57 |
| 2023 November | 60 | 36 | 96 |
| 2023 October | 75 | 24 | 99 |
| 2023 September | 137 | 32 | 169 |
| 2023 August | 62 | 18 | 80 |
| 2023 July | 51 | 15 | 66 |
| 2023 June | 78 | 40 | 118 |
| 2023 May | 61 | 31 | 92 |
| 2023 April | 47 | 14 | 61 |
| 2023 March | 81 | 47 | 128 |
| 2023 February | 76 | 46 | 122 |
| 2023 January | 84 | 26 | 110 |
| 2022 December | 76 | 44 | 120 |
| 2022 November | 93 | 46 | 139 |
| 2022 October | 103 | 53 | 156 |
| 2022 September | 81 | 97 | 178 |
| 2022 August | 62 | 49 | 111 |
| 2022 July | 59 | 55 | 114 |
| 2022 June | 68 | 39 | 107 |
| 2022 May | 65 | 39 | 104 |
| 2022 April | 57 | 45 | 102 |
| 2022 March | 58 | 51 | 109 |
| 2022 February | 60 | 41 | 101 |
| 2022 January | 102 | 40 | 142 |
| 2021 December | 68 | 40 | 108 |
| 2021 November | 60 | 50 | 110 |
| 2021 October | 70 | 59 | 129 |
| 2021 September | 54 | 44 | 98 |
| 2021 August | 49 | 36 | 85 |
| 2021 July | 30 | 18 | 48 |
| 2021 June | 27 | 40 | 67 |
| 2021 May | 40 | 38 | 78 |
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