Scientific Letter
‘Unventilable bronquiolitis’ as symptom of congenital tracheal stenosis
«Bronquiolitis inventilables» como manifestación de estenosis traqueales congénitas
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Typical clinical manifestations include respiratory distress, cyanosis, dysphagia or difficulty with ingestion, and stridor<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a>. In older patients, it may manifest as repeat pneumonias.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Tracheal narrowing may occasionally be seen on chest X-ray, but the diagnostic method of choice is fiberoptic bronchoscopy, while CT and MRI are useful for defining stenotic extension<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a>.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Treatment requires surgical correction<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a>, and the conventional options include laryngotracheal reconstruction, slide tracheoplasty, and partial cricotracheal resection<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8–11</span></a>. Less invasive procedures such as balloon dilatation, implantation of endoluminal stents in the area of stenosis, or laser treatment are also available.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Historically, prognosis depended on the extension of the stenosis, but surgical advances have improved morbidity and mortality in patients with severe involvement<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a>.</p><p id="par0035" class="elsevierStylePara elsevierViewall">We report the cases of 2 patients aged 2 and 4 months who were admitted to the Pediatric Intensive Care Unit (PICU) of our hospital with symptoms of respiratory failure in the context of respiratory syncytial virus (RSV) bronchiolitis. Their ventilatory support requirements increased significantly and were difficult to manage appropriately.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The first patient, who had a post-natal diagnosis of trisomy 21, monitored by cardiology for patent ductus arteriosus and by nephrology for left pelvic ectasia, was admitted to the PICO at 2 months of age for respiratory failure due to RSV bronchiolitis. His initial progress on non-invasive ventilation (NIV) with a combination of helium and oxygen was good, but he later required endotracheal intubation and invasive mechanical ventilation (IMV).</p><p id="par0045" class="elsevierStylePara elsevierViewall">The second patient was a late premature twin (35 + 4 weeks gestation). At 4 months of life he presented respiratory failure due to bronchiolitis caused by RSV, which required admission to the PICU. He initially received NIV, with progressive failure and need for IMV.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Both patients were initially connected to bi-level IMV (intermittent mandatory mode) delivered via a face mask (the first initially in continuous pressure with helium-oxygen) but showed progressive clinical and blood gas deterioration despite support optimization, so intubation and connection to IMV were required. Both patients were difficult to intubate, with resistance to correct endotracheal tube (ETT) advancement due to their small endoluminal caliber.</p><p id="par0055" class="elsevierStylePara elsevierViewall">They presented a predominantly obstructive mixed pattern, requiring IMV in volume control mode, with difficult-to-manage ventilatory status despite optimization of support and intensification of bronchodilator treatment (nebulized salbutamol and ipratropium bromide, corticosteroids, intravenous theophylline and magnesium sulphate, sedation and analgesia, and muscle relaxation). They had very high peak inspiratory pressures (PIP) (up to 90 cmH<span class="elsevierStyleInf">2</span>O), which made it difficult to achieve adequate tidal volumes, despite ventilating without PIP limit, resulting in respiratory acidosis with severe hypercapnia (pCO<span class="elsevierStyleInf">2</span> > 150 mmHg), with moments of intermittent improvement.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In light of their slow progress, the original diagnostic work-up was complemented with fiberoptic bronchoscopy and pulmonary CT angiogram. In the second patient, stenosis was not observed in the first fiberoptic bronchoscopy that was performed immediately after intubation, in view of the significant diagnostic suspicion generated by the difficulty of intubation.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The first patient presented distal tracheal stenosis due to compression of the brachiocephalic artery, with a critical diameter of 1 mm (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The second patient had distal infundibular tracheal stenosis of 90% of the tracheal lumen, 5 cm in length with a critical diameter of 1 mm in the supracarinal region and 4−5 mm in the cervical trachea.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0070" class="elsevierStylePara elsevierViewall">Both patients underwent slide tracheoplasty at the reference hospital for airway diseases. The first patient made good post-surgical progress. The second patient initially underwent slide tracheoplasty of the entire length of the trachea (except the first and second rings), with reimplantation of the left pulmonary branch. He subsequently required several reinterventions (balloon dilations and placement of 2 tracheal endoluminal prostheses). Finally, tracheostomy was performed with subsequent partial tracheal resection due to recurrent peristomal granulomas.</p><p id="par0075" class="elsevierStylePara elsevierViewall">A critical situation in patients with tracheal stenosis is usually triggered by an inflammatory component associated or not with mucous plugs that worsen existing narrowing, as in our patients, due to bronchiolitis caused by RSV. It is often possible to intubate these patients, but difficulty can be encountered when advancing the tube distally to the glottis. Technological developments that offer 3-dimensional airway reconstructions have facilitated the development of “virtual tracheobronchoscopy”, a significant advance in the diagnosis of tracheobronchial anomalies<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">13,14</span></a>. The underlying diagnostic suspicion arises at the time of intubation and usually persists in the presence of a severe obstructive pattern that hinders ventilatory support. However, this severity may be intermittent depending on the depth of ETT fixation relative to the extent of the lesion, as observed in both patients.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Affected patients, when clinically destabilized, usually require IMV for a median duration of 59 days<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a>. Some studies have analyzed various parameters to evaluate predicted survival, and conclude that neither the duration of ventilatory support nor the extent of stenosis measured by dynamic contrast bronchoscopy are good predictive tools<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a>.</p><p id="par0085" class="elsevierStylePara elsevierViewall">In PICU patients with slow-progressing obstructive respiratory disease and difficult ventilatory management, fiberoptic bronchoscopy may be useful to rule out hitherto asymptomatic congenital airway malformations and possible complications of intensive and prolonged respiratory support (granulomas, stenosis, vocal cord paralysis).</p><p id="par0090" class="elsevierStylePara elsevierViewall">As congenital tracheal stenosis is rare, a high degree of suspicion is needed to complete and establish the diagnosis. Management is complex and requires an individualized approach by expert multidisciplinary teams working in reference units. The treatment is surgical, and outcomes can vary depending on the techniques used and the series described.</p><p id="par0095" class="elsevierStylePara elsevierViewall">Ventilatory support of these patients in the PICU is challenging. When the extent of the lesion exceeds the length of the TEE, it causes a severe obstructive pattern, and ventilatory support can be very complicated and intensive.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0100" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interests</span><p id="par0105" class="elsevierStylePara elsevierViewall">None.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of interests" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Baquedano Lobera I, Gil Hernández I, Madurga Revilla P. «Bronquiolitis inventilables» como manifestación de estenosis traqueales congénitas. Arch Bronconeumol. 2021;57:660–661.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1265 "Ancho" => 1500 "Tamanyo" => 192695 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Narrowing of the tracheal lumen where it meets the brachiocephalic artery, affecting mainly the anteroposterior axis, which is reduced to 1.3 mm, with a laterolateral axis of 3.3 mm.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Assessment of children with vascular ring" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 2 | 0 | 2 |
| 2024 October | 42 | 23 | 65 |
| 2024 September | 47 | 18 | 65 |
| 2024 August | 71 | 26 | 97 |
| 2024 July | 47 | 19 | 66 |
| 2024 June | 62 | 24 | 86 |
| 2024 May | 83 | 26 | 109 |
| 2024 April | 49 | 21 | 70 |
| 2024 March | 50 | 17 | 67 |
| 2024 February | 49 | 30 | 79 |
| 2024 January | 40 | 23 | 63 |
| 2023 December | 41 | 28 | 69 |
| 2023 November | 38 | 38 | 76 |
| 2023 October | 58 | 37 | 95 |
| 2023 September | 66 | 33 | 99 |
| 2023 August | 40 | 26 | 66 |
| 2023 July | 46 | 17 | 63 |
| 2023 June | 52 | 21 | 73 |
| 2023 May | 69 | 84 | 153 |
| 2023 April | 50 | 45 | 95 |
| 2023 March | 53 | 25 | 78 |
| 2023 February | 41 | 19 | 60 |
| 2023 January | 44 | 28 | 72 |
| 2022 December | 63 | 26 | 89 |
| 2022 November | 54 | 29 | 83 |
| 2022 October | 68 | 35 | 103 |
| 2022 September | 48 | 25 | 73 |
| 2022 August | 45 | 51 | 96 |
| 2022 July | 51 | 48 | 99 |
| 2022 June | 51 | 45 | 96 |
| 2022 May | 45 | 30 | 75 |
| 2022 April | 69 | 27 | 96 |
| 2022 March | 66 | 44 | 110 |
| 2022 February | 34 | 31 | 65 |
| 2021 November | 1 | 0 | 1 |
| 2021 October | 1 | 0 | 1 |
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