Letter to the Editor
Pulmonary Benign Metastasizing Leiomyoma, a Rare Cause of Pulmonary Nodules
Leiomiomatosis pulmonar benigna metastatizante, una causa excepcional de nódulos pulmonares
Patricia Pérez-Ferrer, Eusebi Chiner
, José Norberto Sancho-Chust, Mar Arlandis
Corresponding author
Servicio de Neumología, Hospital Universitario San Juan de Alicante, San Juan de Alicante, Alicante, Spain
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(B) Chest magnetic resonance image showing an 11<span class="elsevierStyleHsp" style=""></span>cm, lobulated, well-defined heterogeneous left paramediastinal mass, with a solid-cystic component.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pulmonary benign metastasizing leiomyoma (PBML) is a rare disease which occurs in women of child-bearing potential with a history of uterine leiomyoma (UL). Although these tumors are histologically benign, they can metastasize to other organs, such as the lung.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 53-year-old woman with chronic abdominal pain, who was referred after a computed axial tomography (CAT) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) revealed a 6<span class="elsevierStyleHsp" style=""></span>cm mass in the left ovary, multiple bilateral pulmonary nodules, and an 11<span class="elsevierStyleHsp" style=""></span>cm solid-cystic mass in the left paramediastinum. She had a history of hysterectomy for UL 12 years previously; she was a non-smoker and asymptomatic. Lung function, bronchoscopy, and laboratory test results were normal. No uptake was found on PET-CAT, while magnetic resonance imaging (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B) identified a lobulated, well-defined, heterogeneous paramediastinal mass.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Biopsy of a nodule revealed smooth muscle tissue with no atypical cells, low mitotic index, actin/desmin expression, and positive estrogen/progesterone receptors, consistent with leiomyoma. Resection of the pelvic and paramediastinal masses confirmed that both were leiomyomas. The patient received tamoxifen 20<span class="elsevierStyleHsp" style=""></span>mg/day and triptorelin 3.5<span class="elsevierStyleHsp" style=""></span>mg/month, and is currently stable.</p><p id="par0020" class="elsevierStylePara elsevierViewall">UL is the most common gynecological tumor in fertile women. On rare occasions, it grows outside the uterus, when it is called PBML. Around 100 cases have been published since the first report by Steiner in 1939.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">PBML is characterized by multiple extrauterine nodules of smooth muscle tissue, a history of hysterectomy for UL, and a latency period of 3–20 years.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> The most commonly affected organ is the lung, but it may also embed in the pleura, peritoneum, vena cava, or the heart.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Most occurrences are asymptomatic, and it presents as an incidental finding of well-defined multiple pulmonary nodules of varying sizes,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> 70% bilateral, and 17% unilateral. It presents only exceptionally as a solitary nodule.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> More uncommon are the miliary pattern, or cystic or cavitated lesions.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Histological features include actin and desmin expression, positive estrogen and progesterone receptors, and benignity, a low mitotic index, no atypical cells, and no tumor necrosis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> It is produced by independent multifocal proliferations of smooth muscle which respond to hormonal stimulus, or by hematogenous dissemination of an initial UL.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3,4</span></a> Cytogenetic studies suggest a monoclonal origin. Its course is generally indolent, except when the site, size or number of lesions cause complications.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The therapeutic approach to PBML is conservative or surgical, and primary excision is the intervention of choice, whenever possible.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> In our case, the paramediastinal mass was resected after it increased in size, to avoid local complications due to compression of the adjacent mediastinal structures, and to confirm the nature of this large cystic lesion that differed greatly from the other nodules. Resection of these lesions is recommended, in order to avoid complications, such as massive hemoptysis, and to rule out low-grade leiomyosarcoma. Hormone treatment is recommended for inoperable lesions.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">To conclude, PBML should be taken into consideration in women of child-bearing age with multiple pulmonary nodules and history of UL.</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: Pérez-Ferrer P, Chiner E, Sancho-Chust JN, Arlandis M. Leiomiomatosis pulmonar benigna metastatizante, una causa excepcional de nódulos pulmonares. Arch Bronconeumol. 2016;52:226–227.</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" => 569 "Ancho" => 1810 "Tamanyo" => 120040 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest computed tomography, showing a cross-sectional slice of the left paramediastinal mass and a nodule in segment 6 which was biopsied. (B) Chest magnetic resonance image showing an 11<span class="elsevierStyleHsp" style=""></span>cm, lobulated, well-defined heterogeneous left paramediastinal mass, with a solid-cystic component.</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" => "Metastasizing fibroleiomyoma of the uterus: report of a case and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "P.E. 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Cao" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.3892/ol.2015.2878" "Revista" => array:6 [ "tituloSerie" => "Oncol Lett" "fecha" => "2015" "volumen" => "9" "paginaInicial" => "1347" "paginaFinal" => "1350" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25663911" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005200000004/v2_201604010234/S157921291600032X/v2_201604010234/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/15792129/0000005200000004/v2_201604010234/S157921291600032X/v2_201604010234/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S157921291600032X?idApp=UINPBA00003Z" ]
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| 2024 February | 31 | 15 | 46 |
| 2023 March | 5 | 2 | 7 |
| 2023 February | 40 | 15 | 55 |
| 2023 January | 37 | 36 | 73 |
| 2022 December | 40 | 42 | 82 |
| 2022 November | 43 | 24 | 67 |
| 2022 October | 46 | 22 | 68 |
| 2022 September | 31 | 31 | 62 |
| 2022 August | 39 | 37 | 76 |
| 2022 July | 34 | 48 | 82 |
| 2022 June | 24 | 33 | 57 |
| 2022 May | 45 | 22 | 67 |
| 2022 April | 33 | 41 | 74 |
| 2022 March | 40 | 40 | 80 |
| 2022 February | 33 | 29 | 62 |
| 2022 January | 34 | 51 | 85 |
| 2021 December | 26 | 30 | 56 |
| 2021 November | 32 | 38 | 70 |
| 2021 October | 65 | 46 | 111 |
| 2021 September | 31 | 38 | 69 |
| 2021 August | 35 | 32 | 67 |
| 2021 July | 27 | 19 | 46 |
| 2021 June | 35 | 35 | 70 |
| 2021 May | 33 | 32 | 65 |
| 2021 April | 60 | 73 | 133 |
| 2021 March | 34 | 15 | 49 |
| 2021 February | 30 | 25 | 55 |
| 2021 January | 22 | 13 | 35 |
| 2020 December | 36 | 15 | 51 |
| 2020 November | 40 | 15 | 55 |
| 2020 October | 16 | 14 | 30 |
| 2020 September | 26 | 18 | 44 |
| 2020 August | 27 | 18 | 45 |
| 2020 July | 22 | 22 | 44 |
| 2020 June | 19 | 4 | 23 |
| 2020 May | 26 | 20 | 46 |
| 2020 April | 32 | 19 | 51 |
| 2020 March | 14 | 12 | 26 |
| 2020 February | 34 | 18 | 52 |
| 2020 January | 26 | 20 | 46 |
| 2019 December | 26 | 24 | 50 |
| 2019 November | 32 | 22 | 54 |
| 2019 October | 19 | 9 | 28 |
| 2019 September | 28 | 8 | 36 |
| 2019 August | 24 | 16 | 40 |
| 2019 July | 26 | 14 | 40 |
| 2019 June | 15 | 8 | 23 |
| 2019 May | 27 | 15 | 42 |
| 2019 April | 26 | 29 | 55 |
| 2019 March | 35 | 15 | 50 |
| 2019 February | 31 | 15 | 46 |
| 2019 January | 22 | 14 | 36 |
| 2018 December | 28 | 15 | 43 |
| 2018 November | 40 | 18 | 58 |
| 2018 October | 56 | 15 | 71 |
| 2018 September | 18 | 9 | 27 |
| 2018 May | 15 | 0 | 15 |
| 2018 April | 34 | 7 | 41 |
| 2018 March | 14 | 3 | 17 |
| 2018 February | 14 | 4 | 18 |
| 2018 January | 22 | 6 | 28 |
| 2017 December | 23 | 6 | 29 |
| 2017 November | 22 | 13 | 35 |
| 2017 October | 19 | 5 | 24 |
| 2017 September | 17 | 5 | 22 |
| 2017 August | 16 | 4 | 20 |
| 2017 July | 13 | 4 | 17 |
| 2017 June | 27 | 16 | 43 |
| 2017 May | 27 | 11 | 38 |
| 2017 April | 32 | 9 | 41 |
| 2017 March | 14 | 2 | 16 |
| 2017 February | 10 | 5 | 15 |
| 2017 January | 13 | 4 | 17 |
| 2016 December | 27 | 19 | 46 |
| 2016 November | 36 | 23 | 59 |
| 2016 October | 42 | 21 | 63 |
| 2016 September | 26 | 5 | 31 |
| 2016 August | 43 | 14 | 57 |
| 2016 July | 34 | 12 | 46 |
Show all
