Constrictive bronchiolitis, two clinical reports
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"apellidos" => "Mendes" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/X0873215915188459?idApp=UINPBA00004E" "url" => "/08732159/0000002100000003/v0_201604141146/X0873215915188459/v0_201604141147/en/main.assets" ] "en" => array:11 [ "idiomaDefecto" => true "titulo" => "Constrictive bronchiolitis, two clinical reports" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "165" "paginaFinal" => "166" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "D. Madama, A. Silva, S. Freitas, F. Gamboa" "autores" => array:4 [ 0 => array:4 [ "Iniciales" => "D." "apellidos" => "Madama" "email" => array:1 [ 0 => "madama.daniela@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor1" ] ] ] 1 => array:3 [ "Iniciales" => "A." "apellidos" => "Silva" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 2 => array:3 [ "Iniciales" => "S." "apellidos" => "Freitas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 3 => array:3 [ "Iniciales" => "F." 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(A) Chest CT images with cystic and varicose bronchiectasis, as well as peribronchovascular reticulation. (B) Chest CT images confirming the presence of parenchymal densifications, bilateral ground glass opacities and predominance of a mosaic pattern. (C) Photomicrograph illustrating the presence of agglomerates of collagen surrounding the bronchovascular axes with widespread alveolar distension, resulting in concentric narrowing and obliteration. H stain, 100× original magnification. (D) Photomicrograph illustrating the presence of fibroblast proliferation associated with collagen deposition, as well as alveolar and septal rupture and centrilobular emphysema. These features result in constriction of the airway lumen, which is compatible with the definitive diagnosis of constrictive bronchiolitis. H&E stain, 100× original magnification." ] ] ] "textoCompleto" => "<p class="elsevierStylePara">Dear Editor,</p><p class="elsevierStylePara">Bronchiolitis is a generic term that includes a group of disorders with distinct etiologies, characterized by the presence of inflammation in small airways.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a> Constrictive bronchiolitis is a rare entity within this group.</p><p class="elsevierStylePara">The authors describe the case of an obese non-smoker 30-year-old woman with no relevant family history or occupational exposures, who had suffered from persistent dyspnea and wheezing since childhood, especially during recurrent infectious exacerbations. Laboratory tests showed no significant alterations and autoimmunity work-up was negative. Chest X-ray defined a bilateral reticular pattern and CT scan of the thorax revealed cystic and varicose bronchiectasis as well as peribronchovascular reticulation (<a href="#f0005" class="elsevierStyleCrossRefs">Figure 1</a>A). Pulmonary function tests identified a small airways obstruction pattern. The histological examination of a lung surgical biopsy diagnosed constrictive bronchiolitis (<a href="#f0005" class="elsevierStyleCrossRefs">Figure 1</a>C). The patient was started on oral prednisolone (0.5 mg/kg/day) and azithromycin (500 mg three times a week) with clinical improvement.</p><a name="f0005" class="elsevierStyleCrossRefs"></a><p class="elsevierStylePara"><img src="320v21n03-" alt="Radiological and histological features observed in the described clinical cases (Case 1: A and C, Case 2: B and D). (A) Chest CT images with cystic and varicose bronchiectasis, as well as peribronchovascular reticulation. (B) Chest CT images confirming the presence of parenchymal densifications, bilateral ground glass opacities and predominance of a mosaic pattern. (C) Photomicrograph illustrating the presence of agglomerates of collagen surrounding the bronchovascular axes with widespread alveolar distension, resulting in concentric narrowing and obliteration. H stain, 100× original magnification. (D) Photomicrograph illustrating the presence of fibroblast proliferation associated with collagen deposition, as well as alveolar and septal rupture and centrilobular emphysema. These features result in constriction of the airway lumen, which is compatible with the definitive diagnosis of constrictive bronchiolitis. H&E stain, 100× original magnification."></img></p><p class="elsevierStylePara">Figure 1. Radiological and histological features observed in the described clinical cases (Case 1: A and C, Case 2: B and D). (A) Chest CT images with cystic and varicose bronchiectasis, as well as peribronchovascular reticulation. (B) Chest CT images confirming the presence of parenchymal densifications, bilateral ground glass opacities and predominance of a mosaic pattern. (C) Photomicrograph illustrating the presence of agglomerates of collagen surrounding the bronchovascular axes with widespread alveolar distension, resulting in concentric narrowing and obliteration. H stain, 100× original magnification. (D) Photomicrograph illustrating the presence of fibroblast proliferation associated with collagen deposition, as well as alveolar and septal rupture and centrilobular emphysema. These features result in constriction of the airway lumen, which is compatible with the definitive diagnosis of constrictive bronchiolitis. H&E stain, 100× original magnification.</p><p class="elsevierStylePara">The authors also describe the case of an obese non-smoker 70-year-old woman, also without relevant family history or occupational exposures, who was admitted due to bilateral pneumonia with no causing agent identified. After discharge, she continued to have asthma-like symptoms and type 1 respiratory insufficiency which progressively worsened. Blood laboratory workup revealed no abnormalities. Chest X-ray showed bilateral heterogeneous infiltrates. Chest CT confirmed bilateral ground glass opacities with predominance of a mosaic pattern (<a href="#f0005" class="elsevierStyleCrossRefs">Figure 1</a>B). Pulmonary function tests identified small airways obstruction. The patient underwent surgical lung biopsy which revealed lesions of constrictive bronchiolitis (<a href="#f0005" class="elsevierStyleCrossRefs">Figure 1</a>D). She was started on systemic corticosteroids (prednisolone 0.5 mg/kg/day) and azithromycin (500 mg three times a week); there was gradual clinical improvement and recovery from respiratory insufficiency.</p><p class="elsevierStylePara">The term bronchiolitis refers to a number of clinical entities, which are centered in the small airways, and relates to the presence of inflammation and fibrosis predominantly in the membranous bronchioles.<a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a> The cases reported refer to one of these entities, constrictive bronchiolitis. This is a rare condition, difficult to diagnose with nonspecific symptoms and histological features that can be easily taken for other pathologies. It designates inflammation and fibrosis occurring predominantly in membranous and respiratory bronchioles walls and contiguous tissues, sparing distal respiratory bronchioles, with resultant lumen narrowing.<a href="#bib10" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">3</span></a> Histology is characterized by focal changes, which are difficult to read, making diagnosis sometimes problematical.</p><p class="elsevierStylePara">Clinically, patients usually report dyspnea and cough and, like both our patients, have functional airflow limitation. This reflects the effect of bronchiolar lumens concentric narrowing and eventually luminal obliteration.<a href="#bib8" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">1</span></a> It is a chronic, slowly progressive disease.</p><p class="elsevierStylePara">Chest X-ray is often normal or shows signs of hyperinflation or decreased vascular reticulum. Chest CT scan may help diagnosis, sometimes demonstrating the existence of a mosaic pattern, with areas of air-trapping caused by constricted and partially obstructed bronchioles.<a href="#bib11" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">4</span></a> There are also frequently parenchymal densifications and bilateral ground glass opacities.<a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a></p><p class="elsevierStylePara">There are several causes attributed to this entity. Even though it may be idiopathic, most commonly it is secondary to sequelae from childhood infections or inhalation of toxic gases. It can also occur in patients with connective tissue diseases and is a well-known complication of bone and lung transplantation. More recently, it has been associated with ulcerative colitis, proliferation of neuroendocrine cells in the lung or with cystic fibrosis.<a href="#bib12" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">5</span></a></p><p class="elsevierStylePara">Treatment is based on oral systemic corticosteroid therapy. However, it appears that most cases are steroid-resistant, with development of irreversible airway obstruction.<a href="#bib13" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">6</span></a> Association with other immunosuppressants is also controversial, due to the absence of directed studies. Recent trials have also shown that macrolide in the form of azithromycin (250 mg three times a week) may be important in reducing the inflammatory component.<a href="#bib14" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">7</span></a></p><p class="elsevierStylePara">Prognosis is difficult to establish since most of the available studies only enrolled patients with constrictive bronchiolitis associated to organ transplantation. In such patients, the overall mortality rate is 25%.<a href="#bib14" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">7</span></a> However, for 87% of patients who were asymptomatic there was resolution or stabilization of the disease, compared with 38% of those with moderate symptoms and 40% of those with severe symptoms.<a href="#bib9" class="elsevierStyleCrossRefs"><span class="elsevierStyleSup">2</span></a></p><p class="elsevierStylePara">The authors describe two patients with histological diagnosis of constrictive bronchiolitis in order to highlight an uncommon entity that in clinical practice is often misread. Further studies targeting treatment regimens are needed in the near future.</p><a name="sec0005" class="elsevierStyleCrossRefs"></a><span class="elsevierStyleSectionTitle">Conflicts of interest</span><p class="elsevierStylePara">The authors have no conflicts of interest to declare.</p><p class="elsevierStylePara">Corresponding author. madama.daniela@gmail.com</p>" "pdfFichero" => "320v21n03a90418846pdf001.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig1" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "descripcion" => array:1 [ "en" => "Radiological and histological features observed in the described clinical cases (Case 1: A and C, Case 2: B and D). (A) Chest CT images with cystic and varicose bronchiectasis, as well as peribronchovascular reticulation. (B) Chest CT images confirming the presence of parenchymal densifications, bilateral ground glass opacities and predominance of a mosaic pattern. (C) Photomicrograph illustrating the presence of agglomerates of collagen surrounding the bronchovascular axes with widespread alveolar distension, resulting in concentric narrowing and obliteration. H stain, 100× original magnification. (D) Photomicrograph illustrating the presence of fibroblast proliferation associated with collagen deposition, as well as alveolar and septal rupture and centrilobular emphysema. These features result in constriction of the airway lumen, which is compatible with the definitive diagnosis of constrictive bronchiolitis. H&E stain, 100× original magnification." ] ] 1 => array:6 [ "identificador" => "fig2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "descripcion" => array:1 [ "en" => "Radiological and histological features observed in the described clinical cases (Case 1: A and C, Case 2: B and D). (A) Chest CT images with cystic and varicose bronchiectasis, as well as peribronchovascular reticulation. (B) Chest CT images confirming the presence of parenchymal densifications, bilateral ground glass opacities and predominance of a mosaic pattern. (C) Photomicrograph illustrating the presence of agglomerates of collagen surrounding the bronchovascular axes with widespread alveolar distension, resulting in concentric narrowing and obliteration. H stain, 100× original magnification. (D) Photomicrograph illustrating the presence of fibroblast proliferation associated with collagen deposition, as well as alveolar and septal rupture and centrilobular emphysema. These features result in constriction of the airway lumen, which is compatible with the definitive diagnosis of constrictive bronchiolitis. H&E stain, 100× original magnification." ] ] ] "bibliografia" => array:2 [ "titulo" => "Bibliography" "seccion" => array:1 [ 0 => array:1 [ "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib8" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Bronchiolitis – the pathologist's perspective. Proc Am Thorac Soc. 2006; 3:41-7." "contribucion" => array:1 [ 0 => array:3 [ "titulo" => "Bronchiolitis – the pathologist's perspective." 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| 2021 June | 37 | 27 | 64 |
| 2021 May | 41 | 38 | 79 |
| 2021 April | 62 | 71 | 133 |
| 2021 March | 49 | 12 | 61 |
| 2021 February | 64 | 19 | 83 |
| 2021 January | 35 | 10 | 45 |
| 2020 December | 31 | 7 | 38 |
| 2020 November | 44 | 19 | 63 |
| 2020 October | 37 | 20 | 57 |
| 2020 September | 27 | 20 | 47 |
| 2020 August | 41 | 28 | 69 |
| 2020 July | 47 | 20 | 67 |
| 2020 June | 48 | 15 | 63 |
| 2020 May | 34 | 21 | 55 |
| 2020 April | 35 | 15 | 50 |
| 2020 March | 38 | 28 | 66 |
| 2020 February | 28 | 23 | 51 |
| 2020 January | 44 | 14 | 58 |
| 2019 December | 44 | 15 | 59 |
| 2019 November | 36 | 13 | 49 |
| 2019 October | 40 | 27 | 67 |
| 2019 September | 28 | 18 | 46 |
| 2019 August | 104 | 11 | 115 |
| 2019 July | 131 | 19 | 150 |
| 2019 June | 138 | 16 | 154 |
| 2019 May | 118 | 18 | 136 |
| 2019 April | 127 | 29 | 156 |
| 2019 March | 122 | 12 | 134 |
| 2019 February | 116 | 6 | 122 |
| 2019 January | 132 | 23 | 155 |
| 2018 December | 72 | 5 | 77 |
| 2018 November | 24 | 2 | 26 |
| 2018 October | 35 | 10 | 45 |
| 2018 September | 17 | 8 | 25 |
| 2018 August | 38 | 22 | 60 |
| 2018 July | 29 | 17 | 46 |
| 2018 June | 40 | 15 | 55 |
| 2018 May | 56 | 18 | 74 |
| 2018 April | 56 | 24 | 80 |
| 2018 March | 59 | 13 | 72 |
| 2018 February | 19 | 5 | 24 |
| 2018 January | 46 | 13 | 59 |
| 2017 December | 18 | 26 | 44 |
| 2017 November | 19 | 12 | 31 |
| 2017 October | 20 | 15 | 35 |
| 2017 September | 10 | 11 | 21 |
| 2017 August | 20 | 6 | 26 |
| 2017 July | 15 | 8 | 23 |
| 2017 June | 15 | 19 | 34 |
| 2017 May | 23 | 22 | 45 |
| 2017 April | 9 | 12 | 21 |
| 2017 March | 19 | 17 | 36 |
| 2017 February | 9 | 3 | 12 |
| 2017 January | 15 | 5 | 20 |
| 2016 December | 9 | 14 | 23 |
| 2016 November | 9 | 5 | 14 |
| 2016 October | 5 | 2 | 7 |
| 2016 September | 4 | 0 | 4 |
| 2016 August | 4 | 4 | 8 |
| 2016 July | 2 | 13 | 15 |
| 2016 April | 17 | 1 | 18 |
| 2016 March | 26 | 20 | 46 |
| 2016 February | 38 | 26 | 64 |
| 2016 January | 24 | 26 | 50 |
| 2015 December | 27 | 14 | 41 |
| 2015 November | 17 | 20 | 37 |
| 2015 October | 35 | 21 | 56 |
| 2015 September | 71 | 20 | 91 |
| 2015 August | 48 | 35 | 83 |
| 2015 July | 42 | 24 | 66 |
| 2015 June | 60 | 37 | 97 |
| 2015 May | 134 | 103 | 237 |
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