Diffuse cystic lung disease as the primary tomographic manifestation of bronchiolitis: A case series

array:24 ["pii" => "S2531043720300246" "issn" => "25310437" "doi" => "10.1016/j.pulmoe.2020.01.006" "estado" => "S300" "fechaPublicacion" => "2020-11-01" "aid" => "1442" "copyright" => "Sociedade Portuguesa de Pneumologia" "copyrightAnyo" => "2020" "documento" => "simple-article" "crossmark" => 1 "licencia" => "http://creativecommons.org/licenses/by-nc-nd/4.0/" "subdocumento" => "cor" "cita" => "Pulmonol. 2020;26:403-6" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "itemSiguiente" => array:19 [ "pii" => "S2531043720300970" "issn" => "25310437" "doi" => "10.1016/j.pulmoe.2020.04.016" "estado" => "S300" "fechaPublicacion" => "2020-11-01" "aid" => "1484" "copyright" => "Sociedade Portuguesa de Pneumologia" "documento" => "simple-article" "crossmark" => 1 "licencia" => "http://creativecommons.org/licenses/by-nc-nd/4.0/" "subdocumento" => "cor" "cita" => "Pulmonol. 2020;26:406-9" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "en" => array:10 [ "idiomaDefecto" => true "cabecera" => "Letter to the Editor" "titulo" => "A rare case of pulmonary disease combining alpha-1-antitrypsin deficiency and common variable immunodeficiency" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "406" "paginaFinal" => "409" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1140 "Ancho" => 1505 "Tamanyo" => 179217 ] ] "descripcion" => array:1 [ "en" => "Thoracic CT scan showing emphysema in upper lobes and bronchiectasis in both inferior lobes. No abnormal findings were identified on abdominal CT scan: liver has standard size and regular edges." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "C.S. Sousa, V. Teixeira, V. Pereira, R.B. Pinheiro, S. Seixas, N. Martins" "autores" => array:6 [ 0 => array:2 [ "nombre" => "C.S." "apellidos" => "Sousa" ] 1 => array:2 [ "nombre" => "V." "apellidos" => "Teixeira" ] 2 => array:2 [ "nombre" => "V." "apellidos" => "Pereira" ] 3 => array:2 [ "nombre" => "R.B." "apellidos" => "Pinheiro" ] 4 => array:2 [ "nombre" => "S." "apellidos" => "Seixas" ] 5 => array:2 [ "nombre" => "N." "apellidos" => "Martins" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2531043720300970?idApp=UINPBA00004E" "url" => "/25310437/0000002600000006/v1_202011020636/S2531043720300970/v1_202011020636/en/main.assets" ] "itemAnterior" => array:19 [ "pii" => "S2531043719302181" "issn" => "25310437" "doi" => "10.1016/j.pulmoe.2019.12.001" "estado" => "S300" "fechaPublicacion" => "2020-11-01" "aid" => "1431" "copyright" => "Sociedade Portuguesa de Pneumologia" "documento" => "simple-article" "crossmark" => 1 "licencia" => "http://creativecommons.org/licenses/by-nc-nd/4.0/" "subdocumento" => "cor" "cita" => "Pulmonol. 2020;26:400-3" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:2 [ "total" => 134 "formatos" => array:3 [ "EPUB" => 15 "HTML" => 63 "PDF" => 56 ] ] "en" => array:10 [ "idiomaDefecto" => true "cabecera" => "Letter to the Editor" "titulo" => "Cystic tuberculosis: a very unusual aspect of a common disease" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "400" "paginaFinal" => "403" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1302 "Ancho" => 2167 "Tamanyo" => 262599 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0325" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "Axial (A), coronal (B), and sagittal (C) reformatted CT images showing multiple thin-walled cysts and ill-defined centrilobular nodules, mainly in the upper lung zones. The cysts have bizarre shapes and a branching appearance. (D–F) Minimum intensity projection reformatted images in the same planes as A–C better demonstrate the thin-walled bizarre cysts." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Joane Perim, Eduardo Scarlatelli Pimenta, Edson Marchiori" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Joane" "apellidos" => "Perim" ] 1 => array:2 [ "nombre" => "Eduardo Scarlatelli" "apellidos" => "Pimenta" ] 2 => array:2 [ "nombre" => "Edson" "apellidos" => "Marchiori" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2531043719302181?idApp=UINPBA00004E" "url" => "/25310437/0000002600000006/v1_202011020636/S2531043719302181/v1_202011020636/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "Letter to the Editor" "titulo" => "Diffuse cystic lung disease as the primary tomographic manifestation of bronchiolitis: A case series" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "403" "paginaFinal" => "406" ] ] "autores" => array:4 [ 0 => array:3 [ "autoresLista" => "M.R. de Oliveira, O.M. Dias, A.F. Amaral" "autores" => array:3 [ 0 => array:2 [ "nombre" => "M.R." "apellidos" => "de Oliveira" ] 1 => array:2 [ "nombre" => "O.M." "apellidos" => "Dias" ] 2 => array:2 [ "nombre" => "A.F." "apellidos" => "Amaral" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Divisao de Pneumologia, Instituto do Coracao (InCor), Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "autoresLista" => "E.C.T. do Nascimento" "autores" => array:1 [ 0 => array:2 [ "nombre" => "E.C.T." "apellidos" => "do Nascimento" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Patologia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "autoresLista" => "M. Wanderley" "autores" => array:1 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Wanderley" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Radiologia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil" "identificador" => "aff0015" ] ] ] 3 => array:4 [ "autoresLista" => "C.R.R. Carvalho, B.G. Baldi" "autores" => array:2 [ 0 => array:2 [ "nombre" => "C.R.R." "apellidos" => "Carvalho" ] 1 => array:4 [ "nombre" => "B.G." "apellidos" => "Baldi" "email" => array:1 [ 0 => "bruno.guedes2@terra.com.br" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Divisao de Pneumologia, Instituto do Coracao (InCor), Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author at: Dr Eneas de Carvalho Aguiar Avenue, 44, Fifth Floor, Sao Paulo 05403-900, Brazil." ] ] ] ] "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" => 1296 "Ancho" => 1505 "Tamanyo" => 392847 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0075" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "Chest HRCT scans (A and B) and histopathological findings (C and D) of a 38-year-old woman with chronic cellular bronchiolitis and diffuse cystic lung disease. (A) Axial CT image shows diffuse, regular, and thin-walled pulmonary cysts. The quantification of cystic lung lesions is depicted in blue (B). The percentage of the total lung area occupied with cysts is 2.75%. The photomicrographies (C and D) show mild inflammatory mononuclear cells infiltrating some of the bronchiolar walls. There are cystic alveolar changes in the nearby parenchyma tissue. Lymphoid follicles with reactive germinal centers are shown (hematoxylin and eosin stain) (D). Magnifications: C. ×13; D. ×70." ] ] ] "textoCompleto" => "The differential diagnosis of diffuse cystic lung diseases (DCLDs) includes a wide range of etiologies with different underlying pathophysiologic mechanisms.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a> Although morphological features, such as shape, distribution within the lung parenchyma and adjacent structures, and the presence of other pulmonary manifestations may suggest a specific underlying disease, a significant overlap exists between tomographic findings from different etiologies. In these cases, a lung biopsy may be required to establish a diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a>Lymphangioleiomyomatosis (LAM) is a rare slowly progressive neoplastic lung disease, which has a characteristic radiological appearance and affects mainly women of childbearing age. However, in women with regular and thin-walled cysts without extrapulmonary features compatible with LAM and with low levels of serum vascular endothelial growth factor D (VEGF-D), other potential rare etiologies may be included in the differential diagnosis, such as bronchiolitis, and smoking-related DCLDs.<a class="elsevierStyleCrossRefs" href="#bib0015">3–5</a> We present the cases of eight women that were initially suspected with LAM whose histopathological analysis was compatible with bronchiolitis.Among 347 patients with DCLDs followed at our center since 2006, eight (2.3%) had diffuse pulmonary cysts on HRCT and a histological diagnosis of cellular and constrictive bronchiolitis and were assessed in this study. Clinical, functional, tomographic, and histological features were analyzed. Written informed consent was obtained from all patients.Pulmonary function tests adhered to recommended guidelines.<a class="elsevierStyleCrossRefs" href="#bib0030">6–8</a> Computed tomography was performed in a supine position. Quantification of the volume of the cystic lesions was obtained automatically by densitovolumetry using a computer program (Advantage Workstation Thoracic VCAR software; GE Medical Systems, Milwaukee, WS, USA) and by selecting pixels between −1000 and −950 HU on soft tissue filter images. Paraffin blocks of lung tissue were retrieved for histological analysis (hematoxylin and eosin stain). Immunohistochemical staining for smooth muscle actin (SMA) and human melanoma black-45 (HMB-45) antibodies were examined.Clinical and functional features at the time of lung biopsy are summarized in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>. All patients were non-smoking women with a mean age of 43±14 years at diagnosis. The most common symptom was dyspnea (75%). Four patients had a relevant exposure history. No patient had any relevant personal or family medical history. Anti-Sjögren syndrome-related antigen A/Ro or B/La antibodies, the rheumatoid factor, and antinuclear antibodies were negative. Serum inflammatory markers, and immunoglobulins were unremarkable.<elsevierMultimedia ident="tbl0005"></elsevierMultimedia>Mean FEV1 and DLCO were 79±23% predicted, and 76±17% predicted, respectively. An obstructive pattern and reduced DLCO were found in 25% and 38% of patients, respectively.All patients showed diffusely distributed multiple, regular thin-walled cysts on HRCT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Other tomographic patterns were not found. Four patients underwent HRCT to investigate respiratory symptoms while in the remaining, pulmonary cysts were incidentally found during an investigation of abdominal pain or a routine exam of the chest. The median tomographic extent of cysts was 2.51% (interquartile 25%–75%: 0.8%–8.9%). Serum VEGF-D levels were available for only two patients (139 and 407pg/mL).<elsevierMultimedia ident="fig0005"></elsevierMultimedia>All patients underwent a surgical lung biopsy. Histological analysis showed evidence of chronic cellular (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) or constrictive bronchiolitis. Six cases had inflammatory mononuclear cells infiltrating the bronchiolar wall with a patchy distribution and variable intensity. Biopsies of two patients displayed infrequent, poorly formed and randomly distributed non-necrotizing granulomas. Two patients presented bronchiolar fibrosis. One patient had a discreet fibrotic thickening of the submucosa associated with luminal narrowing in some small airways, whereas another patient displayed complete focal obliteration of the bronchiolar lumen with fibrous scar formation. All patients presented with parenchymal cysts characterized by airspace distensions in the centrilobular and subpleural regions. The walls of these lesions contained residual alveolar tissue. Proliferation of immature smooth muscle-like cells was not detected.DCLDs have a broad differential diagnosis. Clinical and tomographic findings combined with a multidisciplinary approach make differentiation of various entities possibile. In our study, we described eight women with DCLDs referred with an initial suspicion of LAM. Following a histopathological analysis they were diagnosed as having cellular or constrictive bronchiolitis, with mild severity, which is a very rare etiology for DCLD. Although suggestive, the tomographic patterns of the diffuse, regular and thin-walled cysts in women are not specific to LAM.<a class="elsevierStyleCrossRefs" href="#bib0010">2,4,5</a> In the absence of definite findings, a pulmonary biopsy is mandatory to diagnose LAM.<a class="elsevierStyleCrossRef" href="#bib0015">3</a> Low availability of serum VEGF-D dosage is a limitation of our study.Some findings on HRCT contribute to establishing the etiology of DCLDs. In pulmonary Langerhans histiocytosis, cysts are usually irregular, predominate in the upper and middle lung zones and may be associated with nodules.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a> In Birt-Hogg-Dubé (BHD) syndrome, cysts are multiple, thin-walled and predominantly basilar and paramediastinal.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a> Cysts in lymphocytic interstitial pneumonia occur in the lower lobes along the peribronchovascular bundle, frequently with ground-glass opacities.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>The typical pathological and immunohistochemical features of LAM were not identified in our study. Histologically, LAM nodules consist of two cellular subpopulations: the spindle cells express SMA and forms the core of the nodules surrounded by epithelioid cells that exhibit immunoreactivity for HMB-45 antibody.<a class="elsevierStyleCrossRef" href="#bib0045">9</a> On histopathology, the morphology of BHD cysts may resemble what was found in our cases. However, we excluded BHD based on clinical and tomographic features.<a class="elsevierStyleCrossRef" href="#bib0050">10</a> Cysts, emphysema, and respiratory bronchiolitis are identified in smoking-related DCLDs, but were not found in our study.<a class="elsevierStyleCrossRef" href="#bib0025">5</a> A combination of the DCLDs and small airway disease is mainly identified in specific conditions, such as Sjögren’s syndrome, and it is thought that chronic small airway damage might lead to cyst formation in these patients. A possible explanation for the lung cyst formation in bronchiolitis is a check-valve obstruction with distal airspace overinflation related to air trapping, distal to the abnormal airways.<a class="elsevierStyleCrossRefs" href="#bib0005">1,2</a>In two cases, histological analysis showed evidence of chronic cellular bronchiolitis associated with infrequent, poorly formed, non-necrotizing granulomas, frequently seen in hypersensitivity pneumonitis (HP). Pulmonary cysts have been described in HP possibly due to partial bronchiolar obstruction.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>Potential etiologies for our patients with bronchiolitis include autoimmune disorders, such as Sjögren syndrome, post viral infections and HP. However, none of the etiologies raised were confirmed.In summary, LAM is a prototypical DCLD with a characteristic appearance on HRCT. However, cystic features alone are inadequate to establish a diagnosis of LAM. In such context, bronchiolitis should be considered in the differential diagnosis of DCLDs.Informed consentPatients gave informed consent for this study.FundingThis research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Conflicts of interestThe authors have no conflicts of interest to declare." "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Informed consent" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-11-24" "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1296 "Ancho" => 1505 "Tamanyo" => 392847 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0075" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "Chest HRCT scans (A and B) and histopathological findings (C and D) of a 38-year-old woman with chronic cellular bronchiolitis and diffuse cystic lung disease. (A) Axial CT image shows diffuse, regular, and thin-walled pulmonary cysts. The quantification of cystic lung lesions is depicted in blue (B). The percentage of the total lung area occupied with cysts is 2.75%. The photomicrographies (C and D) show mild inflammatory mononuclear cells infiltrating some of the bronchiolar walls. There are cystic alveolar changes in the nearby parenchyma tissue. Lymphoid follicles with reactive germinal centers are shown (hematoxylin and eosin stain) (D). Magnifications: C. ×13; D. ×70." ] ] 1 => array:8 [ "identificador" => "tbl0005" "etiqueta" => "Table 1" "tipo" => "MULTIMEDIATABLA" "mostrarFloat" => true "mostrarDisplay" => false "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0080" "detalle" => "Table " "rol" => "short" ] ] "tabla" => array:3 [ "leyenda" => "Values are expressed as mean±SD, median (25th–75th percentiles) or n (%).Definition of abbreviations: BD: bronchodilator; DLCO: carbon monoxide diffusing capacity; FEV1: forced expiratory volume in the first second; FVC: forced vital capacity; mMRC: modified medical research council dyspnea scale; SpO2: oxyhaemoglobin saturation by pulse oximetry." "tablatextoimagen" => array:1 [ 0 => array:2 [ "tabla" => array:1 [ 0 => """ <table border="0" frame="\n \t\t\t\t\tvoid\n \t\t\t\t" class=""><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Female \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">8 (100%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Age at diagnosis (years) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">43±14 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Time between onset of symptoms and diagnosis (years) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2±2 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Current or former smokers \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">0 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Environmental exposure \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">4 (50%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Previous (mold and birds) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (25%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Current (only birds) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (25%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " colspan="2" align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Clinical manifestations at diagnosis</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Dyspnea \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">6 (75%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">mMRC \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (0–1) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Cough \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">4 (50%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Wheezing \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (12.5%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Pneumothorax \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (12.5%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Pleuritic chest pain \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (25%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Xerostomy \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (25%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Xerophtalmia \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (12.5%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Skin lesions \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">0 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">SpO2 on room air (%) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">97±2 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Oxygen use \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">0 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " colspan="2" align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Pulmonary function tests</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">FVC (L) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">3.01±0.73 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">FVC (%predicted) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">88±12 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">FEV1 (L) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2.32±0.85 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">FEV1 (%predicted) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">79±23 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">FEV1/FVC \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">0.73±0.14 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">DLCO (mL/min/mmHg) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">18.2±4.6 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">DLCO (%predicted) \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">76±17 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " colspan="2" align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Functional patterns</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Normal spirometry \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">5 (63%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Obstructive \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (25%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Restrictive \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (12%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Air trapping<a class="elsevierStyleCrossRef" href="#tblfn0005">a</a> \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">2 (29%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Reduced DLCO \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">3 (38%) \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Positive response to BD<a class="elsevierStyleCrossRef" href="#tblfn0010">b</a> \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">1 (17%) \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab2413487.png" ] ] ] "notaPie" => array:2 [ 0 => array:3 [ "identificador" => "tblfn0005" "etiqueta" => "a" "nota" => "Information available for 7 patients." ] 1 => array:3 [ "identificador" => "tblfn0010" "etiqueta" => "b" "nota" => "Information available for 6 patients." ] ] ] "descripcion" => array:1 [ "en" => "Demographic, clinical and functional characteristics (n=8)." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diffuse cystic lung disease. Part II" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "N. Gupta" 1 => "R. Vassallo" 2 => "K.A. Wikenheiser-Brokamp" 3 => "F.X. McCormack" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1164/rccm.201411-2096CI" "Revista" => array:7 [ "tituloSerie" => "Am J Respir Crit Care Med" "fecha" => "2015" "volumen" => "192" "numero" => "1" "paginaInicial" => "17" "paginaFinal" => "29" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25906201" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diffuse cystic lung diseases: differential diagnosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "B.G. Baldi" 1 => "C.R.R. Carvalho" 2 => "O.M. Dias" 3 => "E. Marchiori" 4 => "B. 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Young" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1164/rccm.201709-1965ST" "Revista" => array:7 [ "tituloSerie" => "Am J Respir Crit Care Med" "fecha" => "2017" "volumen" => "196" "numero" => "10" "paginaInicial" => "1337" "paginaFinal" => "1348" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/29140122" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diffuse cystic lung disease of unexplained cause with coexistent small airway disease: a possible causal relationship?" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "C. Rowan" 1 => "D.M. Hansell" 2 => "E. Renzoni" 3 => "T.M. Maher" 4 => "A.U. Wells" 5 => "M.I. Polkey" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1097/PAS.0b013e318237c599" "Revista" => array:7 [ "tituloSerie" => "Am J Surg Pathol" "fecha" => "2012" "volumen" => "36" "numero" => "2" "paginaInicial" => "228" "paginaFinal" => "234" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22082605" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Smoking-related diffuse cystic lung disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "N. Gupta" 1 => "T.V. Colby" 2 => "C.A. Meyer" 3 => "F.X. McCormack" 4 => "K.A. Wikenheiser-Brokamp" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.chest.2018.02.027" "Revista" => array:7 [ "tituloSerie" => "Chest" "fecha" => "2018" "volumen" => "154" "numero" => "2" "paginaInicial" => "e31" "paginaFinal" => "e35" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/30080520" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0030" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Standardisation of the single-breath determination of carbon monoxide uptake in the lung" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "N. Macintyre" 1 => "R.O. Crapo" 2 => "G. Viegi" 3 => "D.C. Johnson" 4 => "C.P. van der Grinten" 5 => "V. 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Letter to the Editor

Diffuse cystic lung disease as the primary tomographic manifestation of bronchiolitis: A case series

M.R. de Oliveira, O.M. Dias, A.F. Amaral

Divisao de Pneumologia, Instituto do Coracao (InCor), Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil

E.C.T. do Nascimento

Departamento de Patologia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil

M. Wanderley

Departamento de Radiologia, Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil

C.R.R. Carvalho, B.G. Baldi

Corresponding author

bruno.guedes2@terra.com.br

Corresponding author at: Dr Eneas de Carvalho Aguiar Avenue, 44, Fifth Floor, Sao Paulo 05403-900, Brazil.

Divisao de Pneumologia, Instituto do Coracao (InCor), Hospital das Clinicas HCFMUSP, Faculdade de Medicina, Universidade de Sao Paulo, Sao Paulo, SP, Brazil

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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The differential diagnosis of diffuse cystic lung diseases &#40;DCLDs&#41; includes a wide range of etiologies with different underlying pathophysiologic mechanisms&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Although morphological features&#44; such as shape&#44; distribution within the lung parenchyma and adjacent structures&#44; and the presence of other pulmonary manifestations may suggest a specific underlying disease&#44; a significant overlap exists between tomographic findings from different etiologies&#46; In these cases&#44; a lung biopsy may be required to establish a diagnosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Lymphangioleiomyomatosis &#40;LAM&#41; is a rare slowly progressive neoplastic lung disease&#44; which has a characteristic radiological appearance and affects mainly women of childbearing age&#46; However&#44; in women with regular and thin-walled cysts without extrapulmonary features compatible with LAM and with low levels of serum vascular endothelial growth factor D &#40;VEGF-D&#41;&#44; other potential rare etiologies may be included in the differential diagnosis&#44; such as bronchiolitis&#44; and smoking-related DCLDs&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> We present the cases of eight women that were initially suspected with LAM whose histopathological analysis was compatible with bronchiolitis&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Among 347 patients with DCLDs followed at our center since 2006&#44; eight &#40;2&#46;3&#37;&#41; had diffuse pulmonary cysts on HRCT and a histological diagnosis of cellular and constrictive bronchiolitis and were assessed in this study&#46; Clinical&#44; functional&#44; tomographic&#44; and histological features were analyzed&#46; Written informed consent was obtained from all patients&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Pulmonary function tests adhered to recommended guidelines&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> Computed tomography was performed in a supine position&#46; Quantification of the volume of the cystic lesions was obtained automatically by densitovolumetry using a computer program &#40;Advantage Workstation Thoracic VCAR software&#59; GE Medical Systems&#44; Milwaukee&#44; WS&#44; USA&#41; and by selecting pixels between &#8722;1000 and &#8722;950 HU on soft tissue filter images&#46; Paraffin blocks of lung tissue were retrieved for histological analysis &#40;hematoxylin and eosin stain&#41;&#46; Immunohistochemical staining for smooth muscle actin &#40;SMA&#41; and human melanoma black-45 &#40;HMB-45&#41; antibodies were examined&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Clinical and functional features at the time of lung biopsy are summarized in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#46; All patients were non-smoking women with a mean age of 43<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>14 years at diagnosis&#46; The most common symptom was dyspnea &#40;75&#37;&#41;&#46; Four patients had a relevant exposure history&#46; No patient had any relevant personal or family medical history&#46; Anti-Sj&#246;gren syndrome-related antigen A&#47;Ro or B&#47;La antibodies&#44; the rheumatoid factor&#44; and antinuclear antibodies were negative&#46; Serum inflammatory markers&#44; and immunoglobulins were unremarkable&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Mean FEV<span class="elsevierStyleInf">1</span> and DLCO were 79<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>23&#37; predicted&#44; and 76<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>17&#37; predicted&#44; respectively&#46; An obstructive pattern and reduced DLCO were found in 25&#37; and 38&#37; of patients&#44; respectively&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">All patients showed diffusely distributed multiple&#44; regular thin-walled cysts on HRCT &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Other tomographic patterns were not found&#46; Four patients underwent HRCT to investigate respiratory symptoms while in the remaining&#44; pulmonary cysts were incidentally found during an investigation of abdominal pain or a routine exam of the chest&#46; The median tomographic extent of cysts was 2&#46;51&#37; &#40;interquartile 25&#37;&#8211;75&#37;&#58; 0&#46;8&#37;&#8211;8&#46;9&#37;&#41;&#46; Serum VEGF-D levels were available for only two patients &#40;139 and 407<span class="elsevierStyleHsp" style=""></span>pg&#47;mL&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">All patients underwent a surgical lung biopsy&#46; Histological analysis showed evidence of chronic cellular &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41; or constrictive bronchiolitis&#46; Six cases had inflammatory mononuclear cells infiltrating the bronchiolar wall with a patchy distribution and variable intensity&#46; Biopsies of two patients displayed infrequent&#44; poorly formed and randomly distributed non-necrotizing granulomas&#46; Two patients presented bronchiolar fibrosis&#46; One patient had a discreet fibrotic thickening of the submucosa associated with luminal narrowing in some small airways&#44; whereas another patient displayed complete focal obliteration of the bronchiolar lumen with fibrous scar formation&#46; All patients presented with parenchymal cysts characterized by airspace distensions in the centrilobular and subpleural regions&#46; The walls of these lesions contained residual alveolar tissue&#46; Proliferation of immature smooth muscle-like cells was not detected&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">DCLDs have a broad differential diagnosis&#46; Clinical and tomographic findings combined with a multidisciplinary approach make differentiation of various entities possibile&#46; In our study&#44; we described eight women with DCLDs referred with an initial suspicion of LAM&#46; Following a histopathological analysis they were diagnosed as having cellular or constrictive bronchiolitis&#44; with mild severity&#44; which is a very rare etiology for DCLD&#46; Although suggestive&#44; the tomographic patterns of the diffuse&#44; regular and thin-walled cysts in women are not specific to LAM&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4&#44;5</span></a> In the absence of definite findings&#44; a pulmonary biopsy is mandatory to diagnose LAM&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Low availability of serum VEGF-D dosage is a limitation of our study&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Some findings on HRCT contribute to establishing the etiology of DCLDs&#46; In pulmonary Langerhans histiocytosis&#44; cysts are usually irregular&#44; predominate in the upper and middle lung zones and may be associated with nodules&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> In Birt-Hogg-Dub&#233; &#40;BHD&#41; syndrome&#44; cysts are multiple&#44; thin-walled and predominantly basilar and paramediastinal&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> Cysts in lymphocytic interstitial pneumonia occur in the lower lobes along the peribronchovascular bundle&#44; frequently with ground-glass opacities&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The typical pathological and immunohistochemical features of LAM were not identified in our study&#46; Histologically&#44; LAM nodules consist of two cellular subpopulations&#58; the spindle cells express SMA and forms the core of the nodules surrounded by epithelioid cells that exhibit immunoreactivity for HMB-45 antibody&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> On histopathology&#44; the morphology of BHD cysts may resemble what was found in our cases&#46; However&#44; we excluded BHD based on clinical and tomographic features&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Cysts&#44; emphysema&#44; and respiratory bronchiolitis are identified in smoking-related DCLDs&#44; but were not found in our study&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> A combination of the DCLDs and small airway disease is mainly identified in specific conditions&#44; such as Sj&#246;gren&#8217;s syndrome&#44; and it is thought that chronic small airway damage might lead to cyst formation in these patients&#46; A possible explanation for the lung cyst formation in bronchiolitis is a check-valve obstruction with distal airspace overinflation related to air trapping&#44; distal to the abnormal airways&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In two cases&#44; histological analysis showed evidence of chronic cellular bronchiolitis associated with infrequent&#44; poorly formed&#44; non-necrotizing granulomas&#44; frequently seen in hypersensitivity pneumonitis &#40;HP&#41;&#46; Pulmonary cysts have been described in HP possibly due to partial bronchiolar obstruction&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Potential etiologies for our patients with bronchiolitis include autoimmune disorders&#44; such as Sj&#246;gren syndrome&#44; post viral infections and HP&#46; However&#44; none of the etiologies raised were confirmed&#46;</p><p id="par0070" class="elsevierStylePara elsevierViewall">In summary&#44; LAM is a prototypical DCLD with a characteristic appearance on HRCT&#46; However&#44; cystic features alone are inadequate to establish a diagnosis of LAM&#46; In such context&#44; bronchiolitis should be considered in the differential diagnosis of DCLDs&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Informed consent</span><p id="par0075" class="elsevierStylePara elsevierViewall">Patients gave informed consent for this study&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0080" class="elsevierStylePara elsevierViewall">This research did not receive any specific grant from funding agencies in the public&#44; commercial&#44; or not-for-profit sectors&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Chest HRCT scans &#40;A and B&#41; and histopathological findings &#40;C and D&#41; of a 38-year-old woman with chronic cellular bronchiolitis and diffuse cystic lung disease&#46; &#40;A&#41; Axial CT image shows diffuse&#44; regular&#44; and thin-walled pulmonary cysts&#46; The quantification of cystic lung lesions is depicted in blue &#40;B&#41;&#46; The percentage of the total lung area occupied with cysts is 2&#46;75&#37;&#46; The photomicrographies &#40;C and D&#41; show mild inflammatory mononuclear cells infiltrating some of the bronchiolar walls&#46; There are cystic alveolar changes in the nearby parenchyma tissue&#46; Lymphoid follicles with reactive germinal centers are shown &#40;hematoxylin and eosin stain&#41; &#40;D&#41;&#46; Magnifications&#58; C&#46; &#215;13&#59; D&#46; &#215;70&#46;</p>"
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          "leyenda" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Values are expressed as mean<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>SD&#44; median &#40;25th&#8211;75th percentiles&#41; or n &#40;&#37;&#41;&#46;</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Definition of abbreviations&#58; BD&#58; bronchodilator&#59; DLCO&#58; carbon monoxide diffusing capacity&#59; FEV<span class="elsevierStyleInf">1</span>&#58; forced expiratory volume in the first second&#59; FVC&#58; forced vital capacity&#59; mMRC&#58; modified medical research council dyspnea scale&#59; SpO<span class="elsevierStyleInf">2</span>&#58; oxyhaemoglobin saturation by pulse oximetry&#46;</p>"
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                  \t\t\t\t">Female&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">8 &#40;100&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t">Age at diagnosis &#40;years&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">43<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>14&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t">Time between onset of symptoms and diagnosis &#40;years&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t">Current or former smokers&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">0&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t">Environmental exposure&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">4 &#40;50&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Previous &#40;mold and birds&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2 &#40;25&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2 &#40;25&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t">Clinical manifestations at diagnosis</td></tr><tr title="table-row"><td class="td-with-role" title="\n
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                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Dyspnea&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">6 &#40;75&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">mMRC&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">1 &#40;0&#8211;1&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Cough&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">4 &#40;50&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Wheezing&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">1 &#40;12&#46;5&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Pneumothorax&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">1 &#40;12&#46;5&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Pleuritic chest pain&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2 &#40;25&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Xerostomy&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2 &#40;25&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Xerophtalmia&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">1 &#40;12&#46;5&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Skin lesions&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">0&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t">SpO<span class="elsevierStyleInf">2</span> on room air &#40;&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">97<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>2&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Oxygen use&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">0&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " colspan="2" align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>FVC &#40;L&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">3&#46;01<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>0&#46;73&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>FVC &#40;&#37;predicted&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">88<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>12&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>FEV<span class="elsevierStyleInf">1</span> &#40;L&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">2&#46;32<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>0&#46;85&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>FEV<span class="elsevierStyleInf">1</span> &#40;&#37;predicted&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">79<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>23&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>FEV<span class="elsevierStyleInf">1</span>&#47;FVC&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">0&#46;73<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>0&#46;14&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>DLCO &#40;mL&#47;min&#47;mmHg&#41;&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">18&#46;2<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>4&#46;6&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>DLCO &#40;&#37;predicted&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">76<span class="elsevierStyleHsp" style=""></span>&#177;<span class="elsevierStyleHsp" style=""></span>17&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " colspan="2" align="left" valign="\n
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                  \t\t\t\t">Functional patterns</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Normal spirometry&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">5 &#40;63&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
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                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Obstructive&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">2 &#40;25&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Restrictive&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">1 &#40;12&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Air trapping<a class="elsevierStyleCrossRef" href="#tblfn0005"><span class="elsevierStyleSup">a</span></a>&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">2 &#40;29&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Reduced DLCO&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttop\n
                  \t\t\t\t">3 &#40;38&#37;&#41;&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t"><span class="elsevierStyleHsp" style=""></span>Positive response to BD<a class="elsevierStyleCrossRef" href="#tblfn0010"><span class="elsevierStyleSup">b</span></a>&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">1 &#40;17&#37;&#41;&nbsp;\t\t\t\t\t\t\n
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Demographic&#44; clinical and functional characteristics &#40;n<span class="elsevierStyleHsp" style=""></span>&#61;<span class="elsevierStyleHsp" style=""></span>8&#41;&#46;</p>"
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            0 => array:3 [
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Diffuse cystic lung disease&#46; Part II"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "N&#46; Gupta"
                            1 => "R&#46; Vassallo"
                            2 => "K&#46;A&#46; Wikenheiser-Brokamp"
                            3 => "F&#46;X&#46; McCormack"
                          ]
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                      ]
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                    0 => array:2 [
                      "doi" => "10.1164/rccm.201411-2096CI"
                      "Revista" => array:7 [
                        "tituloSerie" => "Am J Respir Crit Care Med"
                        "fecha" => "2015"
                        "volumen" => "192"
                        "numero" => "1"
                        "paginaInicial" => "17"
                        "paginaFinal" => "29"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25906201"
                            "web" => "Medline"
                          ]
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              "identificador" => "bib0010"
              "etiqueta" => "2"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Diffuse cystic lung diseases&#58; differential diagnosis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "B&#46;G&#46; Baldi"
                            1 => "C&#46;R&#46;R&#46; Carvalho"
                            2 => "O&#46;M&#46; Dias"
                            3 => "E&#46; Marchiori"
                            4 => "B&#46; Hochhegger"
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                      ]
                    ]
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                    0 => array:2 [
                      "doi" => "10.1590/S1806-37562016000000341"
                      "Revista" => array:7 [
                        "tituloSerie" => "J Bras Pneumol"
                        "fecha" => "2017"
                        "volumen" => "43"
                        "numero" => "2"
                        "paginaInicial" => "140"
                        "paginaFinal" => "149"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28538782"
                            "web" => "Medline"
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              "etiqueta" => "3"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Lymphangioleiomyomatosis diagnosis and management&#58; high-resolution chest computed tomography&#44; transbronchial lung biopsy&#44; and pleural disease management&#46; An Official American Thoracic Society&#47;Japanese Respiratory Society Clinical Practice Guideline"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "N&#46; Gupta"
                            1 => "G&#46;A&#46; Finlay"
                            2 => "R&#46;M&#46; Kotloff"
                            3 => "C&#46; Strange"
                            4 => "K&#46;C&#46; Wilson"
                            5 => "L&#46;R&#46; Young"
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                    0 => array:2 [
                      "doi" => "10.1164/rccm.201709-1965ST"
                      "Revista" => array:7 [
                        "tituloSerie" => "Am J Respir Crit Care Med"
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                        "volumen" => "196"
                        "numero" => "10"
                        "paginaInicial" => "1337"
                        "paginaFinal" => "1348"
                        "link" => array:1 [
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              "etiqueta" => "4"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Diffuse cystic lung disease of unexplained cause with coexistent small airway disease&#58; a possible causal relationship&#63;"
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                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "C&#46; Rowan"
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                            2 => "E&#46; Renzoni"
                            3 => "T&#46;M&#46; Maher"
                            4 => "A&#46;U&#46; Wells"
                            5 => "M&#46;I&#46; Polkey"
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                    0 => array:2 [
                      "doi" => "10.1097/PAS.0b013e318237c599"
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                        "tituloSerie" => "Am J Surg Pathol"
                        "fecha" => "2012"
                        "volumen" => "36"
                        "numero" => "2"
                        "paginaInicial" => "228"
                        "paginaFinal" => "234"
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                            0 => "N&#46; Gupta"
                            1 => "T&#46;V&#46; Colby"
                            2 => "C&#46;A&#46; Meyer"
                            3 => "F&#46;X&#46; McCormack"
                            4 => "K&#46;A&#46; Wikenheiser-Brokamp"
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                      "doi" => "10.1016/j.chest.2018.02.027"
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                        "fecha" => "2018"
                        "volumen" => "154"
                        "numero" => "2"
                        "paginaInicial" => "e31"
                        "paginaFinal" => "e35"
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                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/30080520"
                            "web" => "Medline"
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              "etiqueta" => "6"
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                0 => array:2 [
                  "contribucion" => array:1 [
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                      "titulo" => "Standardisation of the single-breath determination of carbon monoxide uptake in the lung"
                      "autores" => array:1 [
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                          "etal" => true
                          "autores" => array:6 [
                            0 => "N&#46; Macintyre"
                            1 => "R&#46;O&#46; Crapo"
                            2 => "G&#46; Viegi"
                            3 => "D&#46;C&#46; Johnson"
                            4 => "C&#46;P&#46; van der Grinten"
                            5 => "V&#46; Brusasco"
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                    0 => array:2 [
                      "doi" => "10.1183/09031936.05.00034905"
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                        "tituloSerie" => "Eur Respir J"
                        "fecha" => "2005"
                        "volumen" => "26"
                        "numero" => "4"
                        "paginaInicial" => "720"
                        "paginaFinal" => "735"
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                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/16204605"
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Article information

ISSN: 25310437
Original language: English

DOI:10.1016/j.pulmoe.2020.01.006

The statistics are updated each day

Year/Month	Html	Pdf	Total

2024 October	33	29	62
2024 September	40	34	74
2024 August	48	35	83
2024 July	53	30	83
2024 June	47	34	81
2024 May	58	44	102
2024 April	61	34	95
2024 March	51	31	82
2024 February	42	27	69
2024 January	42	29	71
2023 December	36	31	67
2023 November	49	58	107
2023 October	33	32	65
2023 September	35	46	81
2023 August	22	18	40
2023 July	21	26	47
2023 June	27	18	45
2023 May	50	26	76
2023 April	35	19	54
2023 March	60	17	77
2023 February	35	26	61
2023 January	37	21	58
2022 December	59	29	88
2022 November	90	39	129
2022 October	83	42	125
2022 September	41	45	86
2022 August	34	49	83
2022 July	48	58	106
2022 June	28	31	59
2022 May	41	40	81
2022 April	34	39	73
2022 March	38	47	85
2022 February	53	36	89
2022 January	63	53	116
2021 December	43	46	89
2021 November	41	34	75
2021 October	42	59	101
2021 September	37	42	79
2021 August	40	36	76
2021 July	35	26	61
2021 June	51	39	90
2021 May	26	40	66
2021 April	123	140	263
2021 March	62	60	122
2021 February	52	39	91
2021 January	63	37	100
2020 December	72	34	106
2020 November	136	57	193
2020 October	37	30	67
2020 September	56	48	104
2020 August	75	35	110
2020 July	59	28	87
2020 June	35	23	58
2020 May	38	19	57
2020 April	50	9	59
2020 March	36	19	55
2020 February	19	11	30

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