Letter to the Editor
Pulmonary Cyst Associated With Primary Sjögren's Syndrome
Quistes pulmonares asociados a síndrome de Sjögren primario
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"paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "116" "paginaFinal" => "118" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Sandra Masegosa-Casanova, Anne Riveros-Frutos, Juana Sanint, Alejandro Olivé" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Sandra" "apellidos" => "Masegosa-Casanova" "email" => array:1 [ 0 => "zuleya86@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" => "Anne" "apellidos" => "Riveros-Frutos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Juana" "apellidos" => "Sanint" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Alejandro" "apellidos" => "Olivé" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital Universitario San Cecilio, Granada, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Sección de Reumatología, Hospital Universitario Germans Trias i Pujol, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Quistes pulmonares asociados a síndrome de Sjögren primario" ] ] "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" => 2253 "Ancho" => 900 "Tamanyo" => 203273 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">High-resolution computed tomographic study: (a) image showing centrilobular emphysema and cysts; (b) bilateral cystic lung lesions, some with septa in their interior, traction bronchiectasis in both lower lobes, areas of centrilobular and paraseptal emphysema and zones of fibrotic tissue; and (c) image showing multiple bilateral thin-walled pulmonary cysts.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">More than 50% of the patients with primary Sjögren's syndrome (pSS) have extraglandular manifestations, and pulmonary manifestations are observed in 10% of this patient population.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">1</span></a> In 50% of the patients, the involvement is subclinical; however, symptomatic lung disease occurs in 10%. Interstitial lung disease is the most common disorder.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">2–4</span></a> Cystic lung disease is reported much less frequently. This complication is characterized by foci of reduced lung density that have well-defined, thin walls (wall thickness less than 4<span class="elsevierStyleHsp" style=""></span>mm) and a diameter at their largest point of 1<span class="elsevierStyleHsp" style=""></span>cm.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">5</span></a> We report the cases of 3 patients with cystic lung disease associated with pSS. All of them met the 2002 American-European classification criteria for the diagnosis of Sjögren's syndrome (SS).<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">6</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The 3 pSS patients were diagnosed, by means of high-resolution computed tomography (HRCT) of the thorax, as having associated cystic lung disease, together with centrilobular pulmonary emphysema, predominantly in the upper lobes in 2 of the 3 patients. There was no evidence of association with other lung diseases. We did not perform a histological examination, a study that had been described in other reports published up to that time.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Patient no. 1</span><p id="par0015" class="elsevierStylePara elsevierViewall">The first patient was a 74-year-old woman with an 18-year history of pSS. She presented with extraglandular manifestations, such as the vasculitis, polyarteritis nodosa. The most notable laboratory findings were hypergammaglobulinemia and anti-Ro and anti-La antibodies. She was being treated with oral azathioprine at a dose of 50<span class="elsevierStyleHsp" style=""></span>mg/day. During follow-up she developed functional dyspnea grade II. The results of respiratory function tests were normal, and HRCT of the thorax revealed centrilobular pulmonary emphysema, predominantly in upper lobes, and images of thin-walled cysts predominantly in lower lobes (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). The same treatment was maintained, and there were no changes in the pulmonary lesions in subsequent visits.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Patient no. 2</span><p id="par0020" class="elsevierStylePara elsevierViewall">This patient was a 46-year-old woman with a 20-year history of pSS and diagnosed with IgA kappa myeloma. Her extraglandular manifestations were anemia and leukocytoclastic vasculitis, as well as polyarteritis and parotid gland enlargement. She was being treated with hydroxychloroquine (HCQ) 200<span class="elsevierStyleHsp" style=""></span>mg/day, oral pilocarpine 15<span class="elsevierStyleHsp" style=""></span>mg/day and rituximab every 6 months. Laboratory tests revealed elevated acute-phase reactant levels, anemia and lymphopenia, as well as hypergammaglobulinemia with an elevated IgG level and a monoclonal IgA kappa component. Tests for rheumatoid factor (RF), as well as anti-Ro and anti-La antibodies, were positive. During follow-up, she developed dyspnea on heavy exertion. Respiratory function tests revealed a mild restrictive ventilatory defect with a slightly reduced diffusing capacity of the lung for carbon monoxide (DLCO), and HRCT of the thorax showed bilateral cystic lung lesions—some with septa in their interior—predominantly in the bases, traction bronchiectasis in both lower lobes, areas of centrilobular and paraseptal emphysema in upper lobes, together with linear opacities and areas of fibrotic tissue in both lung bases (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b). No modifications were made in her treatment, and there were no changes in the cystic lesions in subsequent visits.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Patient no. 3</span><p id="par0025" class="elsevierStylePara elsevierViewall">The third patient was a 78-year-old woman with a 15-year history of pSS who was also diagnosed with cutaneous amyloidosis. The manifestations were predominantly glandular (xerostomia and xerophthalmia), and there was no extraglandular involvement. She was receiving oral HCQ 200<span class="elsevierStyleHsp" style=""></span>mg/day. The laboratory tests revealed elevated acute-phase reactant levels, polyclonal hypergammaglobulinemia with an elevated IgG level, and positive tests for RF and anti-Ro and anti-La antibodies. During follow-up, she reported dyspnea on moderate exertion and, thus, underwent respiratory function tests, which revealed a mild restrictive ventilatory defect, with a severe reduction in DLCO. Thoracic HRCT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>c) showed multiple bilateral thin-walled pulmonary cysts, with no evidence of nodules or changes in the air space. The findings were the same in subsequent visits.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Cystic lung disease is an uncommon condition that is rarely associated with an autoimmune inflammatory disease. The prevalence of pulmonary cysts in pSS ranges between 12% and 30% of the overall group of pulmonary manifestations. They can be found alone or in association with areas of ground glass attenuation.<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">7–9</span></a> Two hypotheses have been proposed for the formation of these pulmonary cysts, both of which involve the infiltration of immunoglobulin-producing lymphocytes: a valve mechanism that causes cystic changes in the dilated alveolar region as a result of chronic inflammation and lymphoplasmocytic infiltration in peripheral regions; and the destruction of the alveolar wall, in which the direct lymphoplasmocytic infiltration of the alveoli destructs the alveolar structures and creates cystic areas. The most probable scenario involves both mechanisms.<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">7,8,10</span></a> There have been few reports of pulmonary cysts associated with pSS, and the majority of the cases are found to be concomitant with other diseases, such as lymphoid interstitial pneumonitis, lymphoproliferative processes and pulmonary amyloidosis.<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">11,12</span></a> One common characteristic finding is the high erythrocyte sedimentation rate with hypergammaglobulinemia observed in all 3 cases, a direct reflection of the lymphocyte hyperactivity associated with the disease. Compared with the cases in the literature, our patients do not differ with respect to the characteristic radiological findings,<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">10</span></a> although there have been reports of a significant association between seropositivity for anti-SSB/La and clonal lymphoproliferative disorders that we have not identified in our patients.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">7</span></a></p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Patient no. 1" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Patient no. 2" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Patient no. 3" ] 3 => 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: Masegosa-Casanova S, Riveros-Frutos A, Sanint J, Olivé A. Quistes pulmonares asociados a síndrome de Sjögren primario. Reumatol Clin. 2016;12:116–118.</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" => 2253 "Ancho" => 900 "Tamanyo" => 203273 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">High-resolution computed tomographic study: (a) image showing centrilobular emphysema and cysts; (b) bilateral cystic lung lesions, some with septa in their interior, traction bronchiectasis in both lower lobes, areas of centrilobular and paraseptal emphysema and zones of fibrotic tissue; and (c) image showing multiple bilateral thin-walled pulmonary cysts.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:12 [ 0 => array:3 [ "identificador" => "bib0065" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Sjögren's syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "S.E. Carsons" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.rdc.2008.08.012" "Revista" => array:6 [ "tituloSerie" => "Rheum Dis Clin North Am" "fecha" => "2008" "volumen" => "34" "paginaInicial" => "907" "paginaFinal" => "920" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18984411" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0070" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary manifestations of Sjögren's syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "P.Y. Hatron" 1 => "I. Tillie-Leblond" 2 => "D. Launay" 3 => "E. Hachulla" 4 => "A.L. Fauchais" 5 => "B. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 16 | 17 | 33 |
| 2024 October | 68 | 36 | 104 |
| 2024 September | 68 | 26 | 94 |
| 2024 August | 67 | 86 | 153 |
| 2024 July | 57 | 91 | 148 |
| 2024 June | 85 | 120 | 205 |
| 2024 May | 82 | 55 | 137 |
| 2024 April | 76 | 29 | 105 |
| 2024 March | 79 | 27 | 106 |
| 2024 February | 69 | 25 | 94 |
| 2024 January | 71 | 23 | 94 |
| 2023 December | 63 | 28 | 91 |
| 2023 November | 61 | 33 | 94 |
| 2023 October | 48 | 35 | 83 |
| 2023 September | 86 | 46 | 132 |
| 2023 August | 53 | 17 | 70 |
| 2023 July | 46 | 25 | 71 |
| 2023 June | 76 | 29 | 105 |
| 2023 May | 50 | 27 | 77 |
| 2023 April | 46 | 11 | 57 |
| 2023 March | 80 | 31 | 111 |
| 2023 February | 61 | 29 | 90 |
| 2023 January | 60 | 19 | 79 |
| 2022 December | 63 | 36 | 99 |
| 2022 November | 68 | 29 | 97 |
| 2022 October | 52 | 24 | 76 |
| 2022 September | 51 | 30 | 81 |
| 2022 August | 51 | 39 | 90 |
| 2022 July | 47 | 46 | 93 |
| 2022 June | 41 | 39 | 80 |
| 2022 May | 54 | 42 | 96 |
| 2022 April | 50 | 42 | 92 |
| 2022 March | 57 | 50 | 107 |
| 2022 February | 74 | 27 | 101 |
| 2022 January | 80 | 37 | 117 |
| 2021 December | 50 | 49 | 99 |
| 2021 November | 50 | 53 | 103 |
| 2021 October | 57 | 51 | 108 |
| 2021 September | 58 | 37 | 95 |
| 2021 August | 58 | 30 | 88 |
| 2021 July | 37 | 28 | 65 |
| 2021 June | 46 | 26 | 72 |
| 2021 May | 68 | 49 | 117 |
| 2021 April | 105 | 86 | 191 |
| 2021 March | 62 | 19 | 81 |
| 2021 February | 48 | 24 | 72 |
| 2021 January | 36 | 22 | 58 |
| 2020 December | 43 | 20 | 63 |
| 2020 November | 38 | 28 | 66 |
| 2020 October | 12 | 12 | 24 |
| 2020 September | 34 | 25 | 59 |
| 2020 August | 23 | 12 | 35 |
| 2020 July | 20 | 15 | 35 |
| 2020 June | 38 | 16 | 54 |
| 2020 May | 24 | 8 | 32 |
| 2020 April | 19 | 13 | 32 |
| 2020 March | 20 | 6 | 26 |
| 2020 February | 2 | 0 | 2 |
| 2018 May | 5 | 0 | 5 |
| 2018 April | 34 | 6 | 40 |
| 2018 March | 107 | 8 | 115 |
| 2018 February | 18 | 3 | 21 |
| 2018 January | 9 | 4 | 13 |
| 2017 December | 26 | 6 | 32 |
| 2017 November | 16 | 4 | 20 |
| 2017 October | 16 | 5 | 21 |
| 2017 September | 17 | 4 | 21 |
| 2017 August | 17 | 7 | 24 |
| 2017 July | 21 | 11 | 32 |
| 2017 June | 33 | 26 | 59 |
| 2017 May | 32 | 11 | 43 |
| 2017 April | 17 | 7 | 24 |
| 2017 March | 12 | 5 | 17 |
| 2017 February | 9 | 5 | 14 |
| 2017 January | 19 | 6 | 25 |
| 2016 December | 45 | 9 | 54 |
| 2016 November | 33 | 15 | 48 |
| 2016 October | 42 | 12 | 54 |
| 2016 September | 39 | 5 | 44 |
| 2016 August | 44 | 9 | 53 |
| 2016 July | 23 | 10 | 33 |
| 2016 April | 3 | 18 | 21 |
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