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Dichas lesiones son fácilmente sangrantes (C). (D) Imagen de TACAR pulmonar: patrón pulmonar bilateral en vidrio deslustrado y un nódulo de 6<span class="elsevierStyleHsp" style=""></span>mm en el segmento posterior del lóbulo superior derecho. (E) TACAR pulmonar: dilataciones bronquiales cilíndricas con ligero engrosamiento mural de bronquios.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Paula Sánchez-Moreno, Lola Falcón-Neyra, Olaf Neth, Isabel Delgado Pecellín" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Paula" "apellidos" => "Sánchez-Moreno" ] 1 => array:2 [ "nombre" => "Lola" "apellidos" => "Falcón-Neyra" ] 2 => array:2 [ "nombre" => "Olaf" "apellidos" => "Neth" ] 3 => array:2 [ "nombre" => "Isabel" "apellidos" => "Delgado Pecellín" ] ] ] ] ] "idiomaDefecto" => "es" "Traduccion" => array:1 [ "en" => array:9 [ "pii" => "S1579212919301259" "doi" => "10.1016/j.arbr.2019.05.005" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212919301259?idApp=UINPBA00003Z" ] ] "EPUB" => 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true "cabecera" => "<span class="elsevierStyleTextfn">Scientific Letter</span>" "titulo" => "Invasive Mucinous Adenocarcinoma in Congenital Pulmonary Airway Malformation: A Case Report" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "384" "paginaFinal" => "385" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Adenocarcinoma mucinoso invasivo sobre malformación congénita de la vía aérea pulmonar (MCVAP): a propósito de un caso" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 989 "Ancho" => 1400 "Tamanyo" => 197752 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A: High-resolution CT, axial plane. Left lower lobe shows an area of destruction of the pulmonary parenchyma with less vascularity (red circle). The image of the larger cyst shows thickened walls and air-fluid level (arrow). B: Chest angio-CT scan with MIP and MPR reconstructions. An arterial branch is seen emerging from the celiac trunk (arrows) that supplies blood to the cystic mass and to the area of destruction. C: Chest X-ray, lateral projection. Consolidation seen in the left lower lobe (red circle), which in the clinical context of the patient was suggestive of pneumonia. D: Chest CT with intravenous contrast and imaging in arterial phase. The larger cystic lesion remains stable (arrow). Hypodense consolidation containing vessels seen within the CPAM (red circle). E: Macroscopic view of the surgical specimen. A whitish, indurated, condensation (red circle) is seen, extending through the pulmonary parenchyma in the form of nodules of variable size. F: Microscopic view of the lesion. Extensive architectural malformation with cystic areas (black arrows) associated with CPAM. Neoplastic proliferation of atypical mucinous epithelial cells covering the alveolar septa, with areas of pseudopneumonic pattern (red circles).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Alexandre Soler-Perromat, Ivan Vollmer, José Ramírez, Marcelo Sánchez" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Alexandre" "apellidos" => "Soler-Perromat" ] 1 => array:2 [ "nombre" => "Ivan" "apellidos" => "Vollmer" ] 2 => array:2 [ "nombre" => "José" "apellidos" => "Ramírez" ] 3 => array:2 [ "nombre" => "Marcelo" "apellidos" => "Sánchez" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0300289618304617" "doi" => "10.1016/j.arbres.2018.12.001" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289618304617?idApp=UINPBA00003Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212919301272?idApp=UINPBA00003Z" "url" => "/15792129/0000005500000007/v1_201907020952/S1579212919301272/v1_201907020952/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Scientific Letter</span>" "titulo" => "Hemoptysis and Bronchiolitis Obliterans in Children With Recurrent Respiratory Papillomatosis: Adverse Reactions to Nebulized Cidofovir" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "386" "paginaFinal" => "387" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Paula Sánchez-Moreno, Lola Falcón-Neyra, Olaf Neth, Isabel Delgado Pecellín" "autores" => array:4 [ 0 => array:3 [ "nombre" => "Paula" "apellidos" => "Sánchez-Moreno" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 1 => array:4 [ "nombre" => "Lola" "apellidos" => "Falcón-Neyra" "email" => array:1 [ 0 => "l.falcon.neyra@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 2 => array:3 [ "nombre" => "Olaf" "apellidos" => "Neth" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Isabel" "apellidos" => "Delgado Pecellín" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Servicio de Infectología, Reumatología e Inmunología Pediátricas, Hospital Virgen del Rocío, Sevilla, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Instituto de Biomedicina de Sevilla, Sevilla, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Unidad de Fibrosis Quística, Hospital Universitario Virgen del Rocío, Sevilla, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Centro de Investigación Biomédica en Red de Enfermedades Respiratorias (CIBERES), Instituto de Salud Carlos III, Madrid, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hemoptisis y bronquiolitis obliterante en niños con papilomatosis laríngea recurrente: reacciones adversas al cidofovir nebulizado" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1355 "Ancho" => 1665 "Tamanyo" => 202090 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A–C) Fiberoptic bronchoscopy image: pearly skin lesions in the posterior third of the vocal cords and vestibular folds of the glottis, extending to the trachea, causing 90% stenosis of the tracheal lumen. These lesions bleed easily. (D) Lung HRCT image: bilateral pulmonary ground glass pattern and a 6<span class="elsevierStyleHsp" style=""></span>mm nodule in the posterior segment of the right upper lobe. (E) Lung HRCT: cylindrical bronchial dilation with slight thickening of bronchial walls.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Recurrent laryngeal papillomatosis (RLP) is characterized by the recurrent growth of papillomas in the respiratory tract. It is caused by the human papilloma virus (HPV), and serotypes 6 and 11, with low oncogenic risk, account for almost 90% of cases; other serotypes (16, 18, 31, 33) are rare.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">1</span></a> Its incidence is unknown; in the United States, an estimated 4.3:100<span class="elsevierStyleHsp" style=""></span>000 children and 1.8:100<span class="elsevierStyleHsp" style=""></span>000 adults are affected.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">2</span></a> Presentation occurs with a bimodal distribution, with both a juvenile and adult form.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">3</span></a> The juvenile form is the main cause of laryngeal tumors in children and the second cause of spasmodic dysphonia, which is the most frequent guiding symptom,<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">4</span></a> but given its non-specific clinical picture, diagnosis may be delayed for up to a year.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">5</span></a> Infection with HPV 11 and diagnosis before 3 years of age are major risk factors for the development of severe forms, and these factors are associated with more recurrences.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">3</span></a> In the most aggressive forms, the airway may be so severely compromised as to require tracheostomy. The clinical course is unpredictable, and the clinical spectrum varies from progressive and spontaneous remission, through recurrences and endobronchial dissemination of papillomatous lesions<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">6</span></a> to malignant conversion (2%).<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">6</span></a> It is associated with high morbidity and a heavy psychosocial impact on patients and family members, and generates significant healthcare expenditure.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">7</span></a> There is currently no curative treatment.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">3,4</span></a> The mainstay of treatment is surgery with repeated endoscopies to clear obstructions from the airway.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">3,4</span></a> Numerous adjuvant medical treatments have been tested, but with limited success.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">8</span></a> The most commonly used are intralesional cidofovir<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">9</span></a> and systemic interferon.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">10</span></a> The subglottic dissemination of papillomatous lesions worsens prognosis considerably<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">3</span></a> by complicating the surgical approach.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">3</span></a> According to the literature, nebulized cidofovir has been used in cases of distal dissemination,<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">11,12</span></a> and has been well tolerated and shown good pathological response<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">11,12</span></a> (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">We report our experience with the compassionate use of nebulized cidofovir in 2 pediatric patients with RLP with endobronchial dissemination, who responded poorly with significant side effects. Consent for publication of both cases was obtained from the patients’ legal representatives.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Clinical case 1: Girl with a personal history of prematurity and orotracheal intubation at birth. Diagnosed with HPV RLP at 6–9 months of age due to progressive inspiratory stridor. A tracheostomy was placed 3 months after diagnosis. Since then, she has needed monthly surgical interventions for resection of lesions and has received adjuvant treatment with subcutaneous interferon alfa, intralesional cidofovir, oral 3-indolcarbinol, and tetravalent anti-HPV vaccine (Gardasil<span class="elsevierStyleSup">TM</span>). At the age of 3 years, during fiberoptic bronchoscopy, a papillomatous lesion was observed in the right main bronchus. Treatment began with nebulized cidofovir (4<span class="elsevierStyleHsp" style=""></span>ml at a concentration of 10<span class="elsevierStyleHsp" style=""></span>mg/ml, 3 times per week). After administration of the fifth dose, she consulted due to frank hemoptysis and desaturation, requiring hospital admission for control of symptoms. Nebulized cidofovir was suspended, and since then she has not presented any more bleeding episodes.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Clinical case 2: Girl diagnosed with HPV RLP at 11–18 months of age, requiring a tracheostomy to maintain airway patency, which she still requires. Since diagnosis, she has needed fortnightly/monthly surgeries, and has received treatment with intralesional and systemic cidofovir, subcutaneous interferon alpha, and tetravalent anti-HPV vaccine (Gardasil<span class="elsevierStyleSup">TM</span>). At the age of 8 years, she consulted due to progressive respiratory difficulty, and fiberoptic bronchoscopy revealed papillomatous lesions in the trachea (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). We decided to start nebulized cidofovir at a lower concentration, given the previous experience (4<span class="elsevierStyleHsp" style=""></span>ml at a concentration of 5<span class="elsevierStyleHsp" style=""></span>mg/ml, 3 times/week), with good clinical response (minimal residual papillomatous lesions 6 months later). Nine months after starting treatment, she developed constitutional syndrome, hypoxemia, and increased breathing difficulty. HRCT of the lung showed patchy focal areas of ground glass density and bilateral bibasal cylindrical bronchiectasis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), findings that, along with the clinical picture, helped establish the diagnosis of bronchiolitis obliterans organizing pneumonia (BOOP). The nebulized cidofovir was discontinued and corticosteroids started at 2<span class="elsevierStyleHsp" style=""></span>mg/kg, enabling withdrawal of oxygen and leading to clinical improvement. HRCT follow-up of the lung revealed radiological improvement, both in the ground glass pattern and the cylindrical bronchiectasis. Currently, corticosteroids have been suspended, and the patient is receiving aerosol therapy and respiratory physiotherapy for her condition.</p><p id="par0025" class="elsevierStylePara elsevierViewall">In conclusion, the therapeutic management of RLP is a challenge, given the lack of effective therapeutic alternatives, and the significant adverse effects of the available options. In the absence of controlled studies in significant cohorts, the evidence published to date<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">11,12</span></a> does not support the use of nebulized cidofovir in patients with RLP, as its side effects are potentially serious, and its effectiveness is doubtful.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The publication of experience of the efficacy and safety of “off-label” treatments in this rare disease could help optimize therapeutic decision-making in daily clinical practice, improving the management and quality of life of these patients.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sánchez-Moreno P, Falcón-Neyra L, Neth O, Delgado Pecellín I. Hemoptisis y bronquiolitis obliterante en niños con papilomatosis laríngea recurrente: reacciones adversas al cidofovir nebulizado. Arch Bronconeumol. 2019;55:386–387.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1355 "Ancho" => 1665 "Tamanyo" => 202090 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A–C) Fiberoptic bronchoscopy image: pearly skin lesions in the posterior third of the vocal cords and vestibular folds of the glottis, extending to the trachea, causing 90% stenosis of the tracheal lumen. These lesions bleed easily. (D) Lung HRCT image: bilateral pulmonary ground glass pattern and a 6<span class="elsevierStyleHsp" style=""></span>mm nodule in the posterior segment of the right upper lobe. 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Year/Month | Html | Total | |
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2024 November | 3 | 3 | 6 |
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