Letter to the Editor
Effectiveness of rituximab in a case of pleuropulmonary fibroelastosis in a patient with rheumatoid arthritis
Efectividad de rituximab en un caso de fibroelastosis pleuropulmonar en paciente con artritis reumatoide
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(C) Elastic fibres in alveolar walls (Verhoeff ×20). (D) Collagen fibres in the alveolar lumen (Masson’s trichrome ×20).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pleuropulmonary fibroelastosis (PPFE) is a rare interstitial lung disease (ILD), recently included in the ATS/ERS guidelines.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is associated with various processes, including autoimmune and connective tissue diseases, such as rheumatoid arthritis. The incidence of this disease is unknown, having reported around 120 cases in the literature.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The symptomatology is similar to that of other ILDs, differing from them by a typical HRCT image with thinning of the visceral pleura and subpleural fibrosis predominantly in the upper lobes. The diagnosis is one of exclusion, requiring the combination of clinical manifestations, compatible CAT scan and histology by lung biopsy.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The prognosis of this disease is ominous, and the only effective treatment at present is lung transplantation. We report the first case of a patient with rheumatoid arthritis and associated PPFE treated with rituximab, with a good response.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 58-year-old woman, former smoker, with no history of interest, diagnosed with rheumatoid arthritis (RA) at the beginning of 2014. Months later, she developed a left basal pneumonia, for which she received antibiotic treatment and corticosteroids, with improvement; however, the respiratory symptoms returned, and she became corticosteroid dependent.</p><p id="par0015" class="elsevierStylePara elsevierViewall">A chest HRCT was performed, showing biapical fibrotic changes with traction bronchiectasis in both upper lobes with significant volume loss and irregular pleural thickening predominantly in upper lobes, middle lobe, lingula and upper lower lobe segments.</p><p id="par0020" class="elsevierStylePara elsevierViewall">A biopsy was taken by bronchoscopy. Anatomical pathology described fibrosis with marked elastosis affecting the visceral pleural and subpleural parenchyma, extending to septa, surrounding the bronchioles and obliterating the alveoli (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The findings of intra-alveolar fibrosis and septal elastosis are the differential pathological finding that allows the distinction of pleuropulmonary fibroelastosis from other interstitial lung diseases,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> so the diagnosis of PPFE was established in February 2015.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Given the resistance and intolerance to treatment with corticosteroids, leflunomide and azathioprine, it was decided, together with pulmonology, to introduce rituximab, subject to approval by the Hospital Ethics Committee. A total of 7 six-month cycles were scheduled; each of them with 2 doses of 1 g separated by 15 days; of rituximab. Functional tests were normal and remained unchanged. A control HRCT was performed, which reported radiological stability for the PPFE.</p><p id="par0030" class="elsevierStylePara elsevierViewall">A literature review was performed using the MESH terms “Pleuropulmonary Fibroelastosis”, “Pleuroparenchymal Fibroelastosis”, “Interstitial Lung Disease” and “Rituximab”; in the PubMed search engine, between July 2019 and July 2021.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The association of PPFE with different conditions, including autoimmune and connective tissue diseases, has been reported.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Its prognosis is dire, and it is common for patients to deteriorate rapidly, with a survival of between 4 months and 7 years after diagnosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">No treatment has been shown to be effective up to now. Corticosteroids, acetylcysteine and immunosuppressants have been used empirically, with no evidence of improvement, and in many cases lung transplantation<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> has been necessary.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Rituximab is an anti-CD20 molecule with extensive clinical experience in the treatment of ILD associated with autoimmune and connective tissue diseases. For this reason, treatment with this biological drug was tested in our patient. The patient received a total of 7 cycles of rituximab on a six-monthly basis, each with 2 doses of 1 g separated by 15 days, and remained stable, clinically, radiologically and in terms of respiratory function 6 years after the diagnosis of PPFE associated with RA.</p><p id="par0050" class="elsevierStylePara elsevierViewall">CD20 inhibition has been shown to be effective in other interstitial lung diseases secondary to connective tissue diseases, so we conclude that rituximab could be effective in the treatment of PPFE, although more studies will be necessary to confirm this hypothesis.</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: Navidad-Fuentes M, Ramírez de la Torre M, Quirosa-Flores S. Efectividad de rituximab en un caso de fibroelastosis pleuropulmonar en paciente con artritis reumatoide. Med Clin (Barc). 2022;158:e6–e7.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1095 "Ancho" => 2007 "Tamanyo" => 468950 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A and B) Homogeneous subpleural lesion sharply delimited from the healthy parenchyma (H&E A: ×1; B: ×5). (C) Elastic fibres in alveolar walls (Verhoeff ×20). (D) Collagen fibres in the alveolar lumen (Masson’s trichrome ×20).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "An Official American Thoracic Society/European Respiratory Society Statement: update of the International Multidisciplinary Classification of the idiopathic interstitial pneumonias" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "W.D. Travis" 1 => "U. Costabel" 2 => "D.M. Hansell" 3 => "T.E. King" 4 => "D.A. Lynch" 5 => "A.G. 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