Scientific Letter
Intracystic Hemorrhage in a Patient with Pulmonary Cystic Disorder Related to Light-Chain Deposition Disease
Hemorragia intraquística en paciente con afectación pulmonar quística secundaria a enfermedad por depósito de cadenas ligeras
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B) Imagen axial de la TC de tórax (ventana de pulmón) en la que se visualizan múltiples lesiones quísticas confluyentes en la periferia de ambos pulmones. Nótese la presencia de pequeños vasos pulmonares (flechas) que atraviesan las formaciones quísticas. C) Radiografía de tórax en la que se identifica la aparición de un nivel hidroaéreo (flechas) en una lesión quística de la base pulmonar izquierda (asterisco). 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(B) Axial image of chest CT (lung window), showing multiple converging cystic lesions in the periphery of both lungs. Note the presence of small pulmonary vessels (arrows) traversing the cystic formations. (C) Chest radiograph identifying an air-fluid level (arrows) in a cystic lesion in the left lung base (asterisk). (D) Minimum intensity projection (miniIP) axial reconstruction, showing the air-fluid level (arrows) corresponding to bleeding in the interior of a cystic lesion (asterisk) in the left lower lobe.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Light-chain deposition disease (LCDD) is a rare systemic disorder characterized by the accumulation of immunoglobulin light chains in multiple organs, and is associated, in most cases, with multiple myeloma or lymphoproliferative disorders. Unlike amyloidosis, the non-fibrillar deposits are negative on Congo Red staining.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> The organs most often affected are the kidney, the heart, the liver, and the nervous system. LCDD lung involvement is very uncommon. Presentation is generally in the form of converging pulmonary cysts, although some atypical forms occurs, such as nodules or bronchiectasis.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report a case of pulmonary intracystic hemorrhage caused by anticoagulation in a 59-year-old woman, non-smoker, with severe pulmonary cystic involvement related with LCDD. The patient consulted due to progressive dyspnea and chest pain. In addition to her LCDD-related pulmonary cystic disease (she is on the waiting list for a lung transplant), her personal history included a diagnosis 8 years previously of multiple myeloma, and a nephrotic syndrome resistant to steroid treatment related with her LCDD. Chest radiograph (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A) showed multiple cystic images in both lungs, predominantly in the subpleural regions (already known). Computed tomography (CT) angiogram of the chest detected pulmonary thromboembolism (PTE), and large subpleural thin-walled cystic lesions in all lobes (described in previous studies), traversed by pulmonary vessels (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Four weeks after beginning anticoagulation, the patient had an episode of left chest pain and bloody sputum. A chest radiograph revealed an air-fluid level in the left lung base (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). The presence of air-fluid level in the interior of a cystic lesion of the left lower lobe was confirmed on chest CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D). The high density of the intracavitary fluid component suggested recent bleeding. The pulmonary artery branches that surrounded the bleeding cystic formation was then embolized with gelfoam particles, and the patient's symptoms improved.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">LCDD is a rare systemic disease, generally associated with plasma cell dyscrasias or lymphoproliferative syndrome, in which light chains of immunoglobulins are deposited in multiple tissues (particularly the kidney, liver and heart).<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> When the lung is involved, diffuse cystic disease, nodules or bronchiectasis may develop.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> The pulmonary cystic form is the most common type of involvement, featuring diffusely distributed, thin-walled cysts of varying sizes (that tend to converge), with walls lined with pulmonary vessels, and, characteristically, pulmonary vessels traversing the cystic formations<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a> We found no references in the literature to intracystic hemorrhage as a complication of diffuse pulmonary cystic disorder related with LCDD. Nephrotic syndrome is a common occurrence in LCDD (resulting from renal involvement) and increases the risk of thromboembolic disease in these patients, but anticoagulation of thromboembolic events can increase the risk of intracavitary bleeding. In our opinion, it is important to remember in LCDD-related pulmonary cystic disorders that the vessels that characteristically penetrate the cysts are pulmonary vessels (rather than bronchial or other systemic arteries), so if intracystic hemorrhage does occur, embolization of the pulmonary arteries must be considered.</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: Sarasúa LG, Pacios-Blanco R-E, Arrieta P, Chinea-Rodríguez A. Hemorragia intraquística en paciente con afectación pulmonar quística secundaria a enfermedad por depósito de cadenas ligeras. Arch Bronconeumol. 2017;53:285–287.</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" => 1349 "Ancho" => 1622 "Tamanyo" => 222182 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest radiograph, showing multiple subpleural cystic formations (asterisks) in both lungs. (B) Axial image of chest CT (lung window), showing multiple converging cystic lesions in the periphery of both lungs. Note the presence of small pulmonary vessels (arrows) traversing the cystic formations. (C) Chest radiograph identifying an air-fluid level (arrows) in a cystic lesion in the left lung base (asterisk). (D) Minimum intensity projection (miniIP) axial reconstruction, showing the air-fluid level (arrows) corresponding to bleeding in the interior of a cystic lesion (asterisk) in the left lower lobe.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "AJKD atlas of renal pathology: light chain deposition disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A.B. Fogo" 1 => "M.A. Lusco" 2 => "B. Najafian" 3 => "C.E. 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Sabnis" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1097/01.pas.0000213358.18380.d2" "Revista" => array:6 [ "tituloSerie" => "Am J Surg Pathol" "fecha" => "2007" "volumen" => "31" "paginaInicial" => "267" "paginaFinal" => "276" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17255772" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary cystic disorder related to light chain deposition disease" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "M. Colombat" 1 => "M. Stern" 2 => "O. Groussard" 3 => "D. Droz" 4 => "M. Brauner" 5 => "D. Valeyre" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1164/rccm.200510-1620CR" "Revista" => array:6 [ "tituloSerie" => "Am J Respir Crit Care Med" "fecha" => "2006" "volumen" => "173" "paginaInicial" => "777" "paginaFinal" => "780" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/16399989" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary light-chain deposition disease: CT and pathology findings in nine patients" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "S. Sheard" 1 => "A.G. Nicholson" 2 => "L. Edmunds" 3 => "A.C. Wotherspoon" 4 => "D.M. Hansell" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.crad.2015.01.002" "Revista" => array:6 [ "tituloSerie" => "Clin Radiol" "fecha" => "2015" "volumen" => "70" "paginaInicial" => "515" "paginaFinal" => "522" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25659938" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005300000005/v1_201704280925/S1579212917300836/v1_201704280925/en/main.assets" "Apartado" => array:4 [ "identificador" => "49861" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005300000005/v1_201704280925/S1579212917300836/v1_201704280925/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212917300836?idApp=UINPBA00003Z" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 13 | 5 | 18 |
| 2024 October | 87 | 23 | 110 |
| 2024 September | 75 | 15 | 90 |
| 2024 August | 67 | 35 | 102 |
| 2024 July | 60 | 24 | 84 |
| 2024 June | 84 | 35 | 119 |
| 2024 May | 85 | 26 | 111 |
| 2024 April | 38 | 28 | 66 |
| 2024 March | 47 | 15 | 62 |
| 2024 February | 35 | 16 | 51 |
| 2023 March | 23 | 2 | 25 |
| 2023 February | 63 | 27 | 90 |
| 2023 January | 51 | 20 | 71 |
| 2022 December | 76 | 24 | 100 |
| 2022 November | 61 | 19 | 80 |
| 2022 October | 67 | 33 | 100 |
| 2022 September | 45 | 19 | 64 |
| 2022 August | 62 | 37 | 99 |
| 2022 July | 49 | 40 | 89 |
| 2022 June | 68 | 32 | 100 |
| 2022 May | 49 | 24 | 73 |
| 2022 April | 62 | 37 | 99 |
| 2022 March | 87 | 32 | 119 |
| 2022 February | 72 | 25 | 97 |
| 2022 January | 74 | 29 | 103 |
| 2021 December | 40 | 38 | 78 |
| 2021 November | 49 | 42 | 91 |
| 2021 October | 47 | 42 | 89 |
| 2021 September | 40 | 50 | 90 |
| 2021 August | 30 | 25 | 55 |
| 2021 July | 31 | 30 | 61 |
| 2021 June | 34 | 29 | 63 |
| 2021 May | 32 | 39 | 71 |
| 2021 April | 54 | 75 | 129 |
| 2021 March | 52 | 20 | 72 |
| 2021 February | 30 | 23 | 53 |
| 2021 January | 32 | 8 | 40 |
| 2020 December | 31 | 22 | 53 |
| 2020 November | 37 | 12 | 49 |
| 2020 October | 42 | 14 | 56 |
| 2020 September | 69 | 10 | 79 |
| 2020 August | 71 | 33 | 104 |
| 2020 July | 37 | 21 | 58 |
| 2020 June | 27 | 11 | 38 |
| 2020 May | 34 | 14 | 48 |
| 2020 April | 36 | 17 | 53 |
| 2020 March | 21 | 10 | 31 |
| 2020 February | 31 | 14 | 45 |
| 2020 January | 19 | 11 | 30 |
| 2019 December | 42 | 21 | 63 |
| 2019 November | 33 | 14 | 47 |
| 2019 October | 23 | 12 | 35 |
| 2019 September | 33 | 9 | 42 |
| 2019 August | 28 | 8 | 36 |
| 2019 July | 23 | 14 | 37 |
| 2019 June | 27 | 4 | 31 |
| 2019 May | 45 | 14 | 59 |
| 2019 April | 42 | 16 | 58 |
| 2019 March | 35 | 21 | 56 |
| 2019 February | 38 | 21 | 59 |
| 2019 January | 22 | 14 | 36 |
| 2018 December | 31 | 19 | 50 |
| 2018 November | 57 | 24 | 81 |
| 2018 October | 76 | 12 | 88 |
| 2018 September | 22 | 8 | 30 |
| 2018 May | 12 | 1 | 13 |
| 2018 April | 34 | 4 | 38 |
| 2018 March | 15 | 6 | 21 |
| 2018 February | 28 | 14 | 42 |
| 2018 January | 76 | 6 | 82 |
| 2017 December | 73 | 14 | 87 |
| 2017 November | 28 | 7 | 35 |
| 2017 October | 20 | 15 | 35 |
| 2017 September | 22 | 6 | 28 |
| 2017 August | 16 | 21 | 37 |
| 2017 July | 3 | 2 | 5 |
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