Scientific Letter
Pulmonary Artery Leiomyosarcoma Diagnosed by Magnetic Resonance, PET-CT and EBUS-TBNA
Leiomiosarcoma de arteria pulmonar, diagnóstico mediante resonancia magnética, PET-TC y EBUS-TBNA
Laura Romero Francés
, Juan Antonio Royo Prats
Corresponding author
Servicio de Neumología, Hospital Universitario General de Castellón, Castellón de la Plana, Spain
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(b) MRI in T2 showing mildly hyperintense lesion after intravenous gadolinium administration. (c) PET-CT with increased uptake of contrast material at the same region.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Leiomyosarcoma of the pulmonary artery is an entity that is highly difficult to diagnose, due to its low prevalence and similarity to pulmonary embolism (PE). We report the case of a patient in whom the results of magnetic resonance imaging (MRI) and positron emission tomography (PET-CT) changed the diagnosis from suspected PE to pulmonary artery leiomyosarcoma, subsequently confirmed by endobronchial ultrasound transbronchial needle aspiration (EBUS-TBNA).</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 58-year-old man with type 2 diabetes, former smoker of 25 pack-years, underwent transthoracic echocardiogram as part of a kidney donor protocol. This procedure revealed right heart dilation and estimated systolic pulmonary artery pressure (SPAP) of 60<span class="elsevierStyleHsp" style=""></span>mmHg. He presented dyspnea mMRC grade 2, and no other clinical manifestations.</p><p id="par0015" class="elsevierStylePara elsevierViewall">On physical examination, breathing was normal, with basal SpO<span class="elsevierStyleInf">2</span> 96% and blood pressure 140/100<span class="elsevierStyleHsp" style=""></span>mmHg. A II/IV systolic murmur was detected in the aortic area, and the rest of the examination was normal.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Findings suggestive of enlarged right heart were observed on electrocardiogram. Chest radiograph was normal. Pulmonary hypertension due to an interatrial communication was suspected, so we performed a transesophageal echocardiogram (TEE) which ruled out this hypothesis, but revealed a reduced pulmonary artery diameter. Computed tomography angiography (CT angiogram) was requested, which showed occlusion of the left and partial occlusion of the right pulmonary arteries and an increase in the diameter of the pulmonary conus, suggestive of chronic bilateral PE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Although the patient did not have any known risk factors, PE was assumed and anticoagulant therapy was initiated, while additional testing was being performed. A pulmonary scintigraphy was requested, which confirmed the absence of left lung perfusion and no changes in the right lung. Right heart catheterization confirmed severe pulmonary hypertension. However, both D-dimer and Doppler ultrasonography of the lower limbs were negative. Thrombophilia, inflammatory markers, tumor markers, and autoimmunity markers were also negative. After 3 months of anticoagulation, the patient's dyspnea persisted. A follow-up CT angiogram showed an increase in the filling defect in the right pulmonary artery, along with isolated pulmonary infiltrates.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In view of his poor progress, a chest MRI with contrast medium was requested, showing a mediastinal mass with the same intensity as the muscles in T1, and hyperintense in T2 due to contrast uptake, suggestive of leiomyosarcoma of the pulmonary artery (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b). The lesion occupied the entire lumen of the left pulmonary artery and part of the common trunk of the pulmonary artery, with infiltration of the mediastinal fat surrounding the affected vessels, aortopulmonary window and ascending thoracic aorta. Uptake in this region was also observed on whole-body PET-CT (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>c), with no other focal enhancement. The pathological diagnosis of leiomyosarcoma was confirmed by fine needle aspiration (FNA) of the mass by EBUS-TBNA through the nodal station 7, with neoplastic cells expressing vimentin, smooth muscle actin and desmin. The procedure was performed without complications. Due to its extension, the lesion was considered unresectable. The patient died suddenly a few days after the diagnosis was reached.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Leiomyosarcoma of the pulmonary artery is a rare malignant tumor, and just over 100 cases have been reported in the literature.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">1</span></a> Symptoms are often nonspecific and insidious, with signs of right ventricular dysfunction being found on physical examination. The chest radiograph can show pulmonary nodules, dilation of the pulmonary artery, hypoperfusion, and cardiomegaly, but it can also be normal. Transthoracic echocardiogram generally reveals right ventricular dilation and high estimated SPAP; obstruction of the pulmonary artery trunk with no clear cause is sometimes observed. The usefulness of TEE has not been defined.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">2</span></a> CT angiogram detects filling defects in the lumen of the pulmonary artery, but it might not differentiate between a clot and a tumor in the absence of secondary lesions, such as pulmonary nodules or enlarged lymph nodes.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> Differential diagnoses include pulmonary artery arteritis, congenital pulmonary stenosis, chest neoplasms, and chronic PE. Of these, the most common is chronic PE; the other entities are exceptional.</p><p id="par0040" class="elsevierStylePara elsevierViewall">This, along with the lack of symptoms suggestive of a tumor process, makes diagnosis a complex matter. However, lack of response to anticoagulation and the presence of a massive unilateral perfusion defect on the pulmonary scintigraphy should prompt clinicians to reconsider the diagnosis.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The 2 non-invasive tests that can differentiate between clots and masses are MRI with gadolinium contrast and PET-CT: in the MRI, tumor tissue captures contrast material, while a clot does not.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">3</span></a> It has also been demonstrated recently that an increase in 18-fluoro-2-deoxy-D-glucose uptake in a filling defect on PET-CT is suggestive of malignancy.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The definitive diagnosis is reached by a pathology study, generally by autopsy, biopsy, or directly during the surgical act. Diagnosis by EBUS-TBNA, while unusual, has been described previously with good results, and can be proposed as a feasible technique for differentiating between a clot and a tumor.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">5</span></a> The major risk of this technique is bleeding, particularly in patients with pulmonary hypertension, and this risk is increased even further by enlarged bronchial arteries which may be associated with proximal obstruction of the pulmonary arteries.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">6</span></a> However, the probability of bleeding is minimized by using color Doppler combined with real-time ultrasonography to avoid puncturing areas of high blood flow.<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">7,8</span></a> To date, no serious complications have been reported with the use of the EBUS-TBNA in this context.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">9</span></a> For this reason, EBUS-TBNA is being accepted as a new safe, minimally invasive technique for the diagnosis of mediastinal tumors, including tumors of the pulmonary artery. The treatment of choice is complete surgical resection, and if the tumor is unresectable, prognosis is grim.</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: Romero Francés L, Royo Prats JA. Leiomiosarcoma de arteria pulmonar, diagnóstico mediante resonancia magnética, PET-TC y EBUS-TBNA. Arch Bronconeumol. 2017;53:522–523.</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" => 1747 "Ancho" => 950 "Tamanyo" => 123769 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) CT angiogram showing total filling defect of the left pulmonary artery and partial filling defect of the right. (b) MRI in T2 showing mildly hyperintense lesion after intravenous gadolinium administration. (c) PET-CT with increased uptake of contrast material at the same region.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0050" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Leiomiosarcoma primario pulmonar. Informe de un caso y revisión de la literatura" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "M. Murguía-Pérez" 1 => "E. Carrera-Gonzalez" 2 => "A.G. Cruz-Gordillo" 3 => "A. Carrillo-Muñoz" 4 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 38 | 18 | 56 |
| 2024 September | 51 | 16 | 67 |
| 2024 August | 65 | 51 | 116 |
| 2024 July | 54 | 26 | 80 |
| 2024 June | 57 | 40 | 97 |
| 2024 May | 81 | 30 | 111 |
| 2024 April | 38 | 30 | 68 |
| 2024 March | 45 | 18 | 63 |
| 2024 February | 38 | 17 | 55 |
| 2023 March | 11 | 4 | 15 |
| 2023 February | 58 | 20 | 78 |
| 2023 January | 25 | 36 | 61 |
| 2022 December | 64 | 43 | 107 |
| 2022 November | 49 | 25 | 74 |
| 2022 October | 64 | 37 | 101 |
| 2022 September | 34 | 30 | 64 |
| 2022 August | 72 | 58 | 130 |
| 2022 July | 40 | 37 | 77 |
| 2022 June | 40 | 35 | 75 |
| 2022 May | 55 | 40 | 95 |
| 2022 April | 66 | 35 | 101 |
| 2022 March | 79 | 41 | 120 |
| 2022 February | 86 | 32 | 118 |
| 2022 January | 79 | 46 | 125 |
| 2021 December | 44 | 62 | 106 |
| 2021 November | 57 | 64 | 121 |
| 2021 October | 45 | 49 | 94 |
| 2021 September | 29 | 43 | 72 |
| 2021 August | 32 | 37 | 69 |
| 2021 July | 22 | 26 | 48 |
| 2021 June | 40 | 35 | 75 |
| 2021 May | 64 | 44 | 108 |
| 2021 April | 70 | 91 | 161 |
| 2021 March | 41 | 26 | 67 |
| 2021 February | 30 | 32 | 62 |
| 2021 January | 24 | 27 | 51 |
| 2020 December | 31 | 29 | 60 |
| 2020 November | 32 | 20 | 52 |
| 2020 October | 20 | 19 | 39 |
| 2020 September | 19 | 18 | 37 |
| 2020 August | 21 | 12 | 33 |
| 2020 July | 29 | 24 | 53 |
| 2020 June | 25 | 7 | 32 |
| 2020 May | 27 | 16 | 43 |
| 2020 April | 20 | 21 | 41 |
| 2020 March | 24 | 15 | 39 |
| 2020 February | 28 | 19 | 47 |
| 2020 January | 19 | 20 | 39 |
| 2019 December | 23 | 30 | 53 |
| 2019 November | 29 | 22 | 51 |
| 2019 October | 20 | 18 | 38 |
| 2019 September | 67 | 30 | 97 |
| 2019 August | 21 | 18 | 39 |
| 2019 July | 17 | 17 | 34 |
| 2019 June | 14 | 10 | 24 |
| 2019 May | 25 | 12 | 37 |
| 2019 April | 40 | 24 | 64 |
| 2019 March | 31 | 22 | 53 |
| 2019 February | 27 | 19 | 46 |
| 2019 January | 24 | 23 | 47 |
| 2018 December | 18 | 17 | 35 |
| 2018 November | 54 | 17 | 71 |
| 2018 October | 101 | 26 | 127 |
| 2018 September | 49 | 10 | 59 |
| 2018 May | 42 | 0 | 42 |
| 2018 April | 34 | 3 | 37 |
| 2018 March | 75 | 4 | 79 |
| 2018 February | 37 | 9 | 46 |
| 2018 January | 53 | 11 | 64 |
| 2017 December | 66 | 14 | 80 |
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