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Clinical Image
Resolution of Thoracic and Abdominal Lymphangioleiomyomas in a Patient With Lymphangioleiomyomatosis Treated With Sirolimus
Resolución de los linfangioleiomiomas torácicos y abdominales en una paciente con linfangioleiomiomatosis tratada con sirolimus
Luis Cabezaa, María Ángeles Ruiz-Cobosb, Álvaro Casanovab,
Corresponding author
acasanova1977@yahoo.es

Corresponding author.
a Servicio de Medicina Interna, Hospital Universitario del Henares, Coslada, Madrid, Spain
b Servicio de Neumología, Hospital Universitario del Henares, Coslada, Madrid, Spain
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        "titulo" => "Resoluci&#243;n de los linfangioleiomiomas tor&#225;cicos y abdominales en una paciente con linfangioleiomiomatosis tratada con sirolimus"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Chest computed axial tomography &#40;CAT&#41; &#40;A&#41; showing a right paravertebral cystic lesion 3&#46;2<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>1&#46;9<span class="elsevierStyleHsp" style=""></span>cm&#44; consistent with retrocrural lymphangioleiomyoma&#46; Abdominal CAT &#40;B&#41; showing tumor 6&#46;8<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;9<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>5&#46;1<span class="elsevierStyleHsp" style=""></span>cm of retroperitoneal cystic appearance&#44; located in the left para-aortic region&#44; extending to the pelvis&#44; consistent with abdominal lymphangioleiomyoma&#46; Chest-abdominal CAT one year later &#40;C and D&#41; showing practically complete resolution of lesions&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 53-year-old woman was diagnosed with lymphangioleiomyomatosis &#40;LAM&#41; in 2010&#44; presenting diffuse pulmonary involvement with multiple fine walled cysts&#44; right retrocrural cystic lymphangioleiomyomas&#44; and multiple retroperitoneal tumors of cystic appearance&#44; consistent with abdominal lymphangioleiomyomas&#44; the largest being 6&#46;8<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;9<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>5&#46;1 in diameter &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a> A and B&#41;&#46; Lung function on diagnosis showed forced vital capacity &#40;FVC&#41; 3300<span class="elsevierStyleHsp" style=""></span>ml &#40;98&#37;&#41;&#44; forced expiratory volume in 1 second &#40;FEV<span class="elsevierStyleInf">1</span>&#41; 2370<span class="elsevierStyleHsp" style=""></span>ml &#40;82&#37;&#41; and an FEV<span class="elsevierStyleInf">1</span>&#47;FVC ratio 69&#37;&#46; Carbon monoxide diffusing capacity &#40;DLCO&#41; was 89&#37;&#46; She reported symptoms of dyspnea on heavy exertion and intermittent episodes of predominantly catamenial chest and abdominal pain&#46; After 2 years of follow-up&#44; lung function had significantly worsened&#44; with FEV<span class="elsevierStyleInf">1</span> 2040<span class="elsevierStyleHsp" style=""></span>ml &#40;71&#37;&#41; and DLCO 72&#37;&#44; increased basal dyspnea&#44; and catamenial symptoms persisted&#46; In view of her clinical and functional progression&#44; treatment was started with the mammalian target of rapamycin &#40;m-TOR&#41; inhibitor sirolimus at a dose of 2<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">1</span></a> One year after beginning treatment&#44; a follow-up radiological examination showed practically complete resolution of the thoracic and abdominal lymphangioleiomyomas<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">2</span></a> &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C and D&#41;&#46; Clinical improvement and stabilized lung function were observed&#44; with FEV<span class="elsevierStyleInf">1</span> 1980<span class="elsevierStyleHsp" style=""></span>ml &#40;70&#37;&#41; and DLCO 82&#37;&#46; This image shows the beneficial effect of sirolimus in reducing the size of the thoracic and abdominal lymphangioleiomyomas&#44; and in stabilizing lung function in patients with LAM&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Cabeza Osorio L&#44; Ruiz Cobos M&#193;&#44; Casanova Espinosa &#193;&#46; Resoluci&#243;n de los linfangioleiomiomas tor&#225;cicos y abdominales en una paciente con linfangioleiomiomatosis tratada con sirolimus&#46; Arch Bronconeumol&#46; 2016&#59;52&#58;329&#8211;330&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Chest computed axial tomography &#40;CAT&#41; &#40;A&#41; showing a right paravertebral cystic lesion 3&#46;2<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>1&#46;9<span class="elsevierStyleHsp" style=""></span>cm&#44; consistent with retrocrural lymphangioleiomyoma&#46; Abdominal CAT &#40;B&#41; showing tumor 6&#46;8<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>3&#46;9<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>5&#46;1<span class="elsevierStyleHsp" style=""></span>cm of retroperitoneal cystic appearance&#44; located in the left para-aortic region&#44; extending to the pelvis&#44; consistent with abdominal lymphangioleiomyoma&#46; Chest-abdominal CAT one year later &#40;C and D&#41; showing practically complete resolution of lesions&#46;</p>"
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                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Efficacy and safety of sirolimus in lymphangioleiomyomatosis"
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                          "colaboracion" => "National Institutes of Health Rare Lung Diseases Consortium&#59; MILES Trial Group"
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                        "volumen" => "364"
                        "paginaInicial" => "1595"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Everolimus treatment of abdominal lymphangioleiomyoma in five women with sporadic lymphangioleiomyomatosis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "A&#46;M&#46; Mohammadieh"
                            1 => "S&#46;D&#46; Bowler"
                            2 => "E&#46;J&#46; Silverstone"
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                      "Revista" => array:6 [
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                        "volumen" => "199"
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                        "paginaFinal" => "123"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23879512"
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ISSN: 15792129
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