Scientific Letter
The Utility of Diaphragmatic Ultrasound in the Radiological Diagnosis of Systemic Lupus Erythymatosus Patients With Shrinking Lung Syndrome
Diagnóstico radiológico en el shrinking lung syndrome en pacientes con lupus eritematoso sistémico. Utilidad de la ecografía diafragmática
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"laurix_dr@yahoo.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "José Luis" "apellidos" => "Callejas Rubio" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "José Luis" "apellidos" => "Martín-Rodríguez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Radiodiagnóstico, Complejo Hospitalario Universitario de Granada, Granada, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Medicina Interna, Complejo Hospitalario Universitario de Granada, Granada, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Diagnóstico radiológico en el <span class="elsevierStyleItalic">shrinking lung syndrome</span> en pacientes con lupus eritematoso sistémico. Utilidad de la ecografía diafragmática" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 30-year-old woman, native of Bolivia, with no cardiovascular risk factors or toxic habits, diagnosed in December 2012 with systemic lupus erythematosus (SLE) and Sjögren's syndrome. She was receiving treatment with methotrexate, prednisone, and hydroxychloroquine.</p><p id="par0010" class="elsevierStylePara elsevierViewall">She attended the systemic disease clinic in November 2015 due to dyspnea on moderate exertion, accompanied by orthopnea with no associated clinical evidence of infectious disease. Of note on examination were tachypnea and tachycardia with no tolerance to the decubitus position. She was hospitalized for further examinations and treatment.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Additional tests produced the following results:<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">-</span><p id="par0020" class="elsevierStylePara elsevierViewall">Clinical laboratory tests revealed anemia with leukopenia and mild thrombocytosis, and no other changes.</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">-</span><p id="par0025" class="elsevierStylePara elsevierViewall">Lung function tests showed a restrictive pattern and her maximum inspiratory and expiratory pressures were low, particularly maximum inspiratory pressure at 18.4% predicted value.</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">-</span><p id="par0030" class="elsevierStylePara elsevierViewall">Tests to detect heart disease were normal, including electrocardiogram, echocardiography, pro-BNP values, and enzymes for myocardial insult.</p></li></ul></p><p id="par0035" class="elsevierStylePara elsevierViewall">The following radiological tests were performed:<ul class="elsevierStyleList" id="lis0010"><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">1.</span><p id="par0040" class="elsevierStylePara elsevierViewall">Chest radiograph showing elevation of both hemidiaphragms, with no other significant changes.</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">2.</span><p id="par0045" class="elsevierStylePara elsevierViewall">High-resolution computed tomography (HRCT), which showed laminar atelectasis in the right middle and lower lobe.</p></li><li class="elsevierStyleListItem" id="lsti0030"><span class="elsevierStyleLabel">3.</span><p id="par0050" class="elsevierStylePara elsevierViewall">CT-angiogram revealed no evidence of acute or chronic pulmonary thromboembolism.</p></li><li class="elsevierStyleListItem" id="lsti0035"><span class="elsevierStyleLabel">4.</span><p id="par0055" class="elsevierStylePara elsevierViewall">Initial ultrasonography of the chest and diaphragm showed limited diaphragmatic amplitude, both at rest and during deep breathing and voluntary sniff maneuvers, reduced inspiratory time and diaphragmatic cycle, and increased diaphragmatic contraction speed. <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a> lists these parameters on admission and during the follow-up performed 3 months after discharge.</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia></li></ul></p><p id="par0060" class="elsevierStylePara elsevierViewall">Based on these findings, shrinking lung syndrome in a patient with SLE and Sjögren's syndrome was diagnosed. Treatment began with theophylline, salbutamol, and high-dose prednisone. Clinical and ultrasound findings confirmed the patient's good progress, and she was discharged after 15 days of hospitalization.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Lung involvement in SLE is very common, and can occur in up to 60%–80% of cases,<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">1–3</span></a> often in the form of pleurisy with or without pleural effusion, pneumonia, interstitial fibrosis, acute lupus pneumonitis or pulmonary hypertension.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">4</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">A rarer, less common form of lung involvement in SLE is shrinking lung syndrome (SLS): less than 100 cases have been reported in the literature, and the prevalence among lupus patients is estimated to be less than 1%,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> although an increasing number of authors are now claiming that this entity is underdiagnosed in mild cases.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">6</span></a> The first authors to describe this syndrome were Hoffbrand and Beck in 1965,<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">7</span></a> in a study of 24 patients with lupus, 8 of whom developed unexplained dyspnea. They found that all patients had progressive reduction of lung volumes, and a restrictive ventilatory pattern on spirometry, associated with loss of ventilated lung volume on chest radiograph, leading the authors to propose the term “shrinking lung syndrome”.</p><p id="par0075" class="elsevierStylePara elsevierViewall">This syndrome is generally diagnosed 4 years after onset of SLE,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> although cases have been published in which SLS was the first respiratory manifestation of the disease.<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">8–10</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">The SLS triad is formed by elevated hemidiaphragms, dyspnea with normal lung parenchyma and restrictive pattern on spirometry.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Dyspnea with chest pain is the most frequent complaint among SLS patients, along with orthopnea and intolerance to the decubitus position.</p><p id="par0090" class="elsevierStylePara elsevierViewall">The causes of the SLS are not entirely clear, although a number of hypotheses have been put forward; for example, the syndrome is caused by secondary microatelectasias due to a pulmonary surfactant deficiency, or it is caused by lupus myopathy due to T-cells infiltrating the diaphragm and the muscles of the chest wall.<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">11</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">Diagnosis is derived from clinical suspicion, lung function tests showing a restrictive pattern, and radiological tests ruling out other diseases.</p><p id="par0100" class="elsevierStylePara elsevierViewall">Treatment is not well established, but immunosuppressive drugs are the most widely used and prognosis is generally favorable.</p><p id="par0105" class="elsevierStylePara elsevierViewall">Our case meets the criteria of SLS that have been described to date, since this was a patient diagnosed 3 years previously with SLE<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> who had Sjögren's syndrome with anti-Ro<span class="elsevierStyleSup">+</span> antibodies that are often associated with the presence of SLS. Our patient's clinical manifestations were typical, with dyspnea on exertion, chest pain, and orthopnea with intolerance to decubitus position, possibly related with weak respiratory muscles. To reach the presumptive diagnosis, other causes of dyspnea in lupus patients must be ruled out: the diagnosis of SLS is initially reached by exclusion of other entities.</p><p id="par0110" class="elsevierStylePara elsevierViewall">In this respect, we would like to highlight the usefulness of ultrasonography of the diaphragm and the chest when SLS is suspected.<a class="elsevierStyleCrossRefs" href="#bib0135"><span class="elsevierStyleSup">12–15</span></a> This is a relatively simple rapid and non-invasive examination that can be performed at the patient's bedside, and only minimal collaboration is needed to perform forced inspirations when indicated. It provides real-time dynamic data on diaphragmatic function that cannot be obtained from other radiological tests, and as it involves no radiation, it is the procedure of choice in pregnant women and children. Because ultrasonography provides information on diaphragm mobility and excursion, and quantifies diaphragmatic contraction speed, duration of the breathing cycle and inspiratory time, we believe it can replace the fluoroscopic sniff test, the gold standard for evaluating diaphragm function. The objective parameters obtained with ultrasonography help establish the clinical diagnosis and assess the functional response of the diaphragm to treatment, and the study can also be used to explore the pulmonary parenchyma, yielding data suggestive of atelectasis or condensation, and the presence of serositis in the form of pleural or pericardial effusion, common in patients with lupus.</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: Díaz Rubia L, Callejas Rubio JL, Martín-Rodríguez JL. Diagnóstico radiológico en el <span class="elsevierStyleItalic">shrinking lung syndrome</span> en pacientes con lupus eritematoso sistémico. Utilidad de la ecografía diafragmática. Arch Bronconeumol. 2017;53:702–703.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "tbl0005" "etiqueta" => "Table 1" "tipo" => "MULTIMEDIATABLA" "mostrarFloat" => true "mostrarDisplay" => false "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at1" "detalle" => "Table " "rol" => "short" ] ] "tabla" => array:1 [ "tablatextoimagen" => array:1 [ 0 => array:2 [ "tabla" => array:1 [ 0 => """ <table border="0" frame="\n \t\t\t\t\tvoid\n \t\t\t\t" class=""><thead title="thead"><tr title="table-row"><th class="td" title="table-head " colspan="2" align="left" valign="top" scope="col" style="border-bottom: 2px solid black">M-Mode Ultrasonography</th><th class="td" title="table-head " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Normal \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Pre-Treatment \t\t\t\t\t\t\n \t\t\t\t</th><th class="td" title="table-head " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Post-Treatment \t\t\t\t\t\t\n \t\t\t\t</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td" title="table-entry " rowspan="3" align="left" valign="top">Diaphragmatic excursion (cm)</td><td class="td" title="table-entry " align="left" valign="top">Rest \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.6±0.3 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.3±0.1 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.6±0.2 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Deep breathing \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">5.7±1.0 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.4±0.5 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">2.5±0.6 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Voluntary sniff \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">2.6±0.5 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.6±0.2 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.1±0.4 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry " colspan="2" align="left" valign="top">Speed of diaphragmatic contraction (cm/s)</td><td class="td" title="table-entry " align="char" valign="top">1.3±0.4 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">4.0±0.5 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">2.3±0.4 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry " colspan="2" align="left" valign="top">Inspiratory time (s)</td><td class="td" title="table-entry " align="char" valign="top">1.6±0.5 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">0.4±0.2 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.1±0.3 \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="table-entry " colspan="2" align="left" valign="top">Diaphragmatic cycle length (s)</td><td class="td" title="table-entry " align="char" valign="top">4.5±1.1 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">1.6±0.8 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="char" valign="top">3.0±1.0 \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab1605803.png" ] ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Chest and Diaphragm Ultrasonography Values in our Patient Before and After Treatment.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0080" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Diaphragm function and lung involvement in systemic lupus erythematosus" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "C.J. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 5 | 8 |
| 2024 October | 54 | 73 | 127 |
| 2024 September | 51 | 83 | 134 |
| 2024 August | 76 | 118 | 194 |
| 2024 July | 73 | 89 | 162 |
| 2024 June | 63 | 57 | 120 |
| 2024 May | 90 | 55 | 145 |
| 2024 April | 64 | 52 | 116 |
| 2024 March | 55 | 55 | 110 |
| 2024 February | 32 | 55 | 87 |
| 2023 May | 1 | 2 | 3 |
| 2023 March | 6 | 12 | 18 |
| 2023 February | 42 | 47 | 89 |
| 2023 January | 26 | 59 | 85 |
| 2022 December | 59 | 51 | 110 |
| 2022 November | 58 | 68 | 126 |
| 2022 October | 55 | 34 | 89 |
| 2022 September | 36 | 39 | 75 |
| 2022 August | 37 | 53 | 90 |
| 2022 July | 40 | 49 | 89 |
| 2022 June | 30 | 41 | 71 |
| 2022 May | 53 | 53 | 106 |
| 2022 April | 46 | 31 | 77 |
| 2022 March | 32 | 48 | 80 |
| 2022 February | 25 | 25 | 50 |
| 2022 January | 37 | 46 | 83 |
| 2021 December | 51 | 52 | 103 |
| 2021 November | 37 | 45 | 82 |
| 2021 October | 41 | 53 | 94 |
| 2021 September | 29 | 54 | 83 |
| 2021 August | 23 | 39 | 62 |
| 2021 July | 32 | 37 | 69 |
| 2021 June | 46 | 33 | 79 |
| 2021 May | 44 | 48 | 92 |
| 2021 April | 116 | 90 | 206 |
| 2021 March | 52 | 22 | 74 |
| 2021 February | 30 | 26 | 56 |
| 2021 January | 50 | 6 | 56 |
| 2020 December | 37 | 30 | 67 |
| 2020 November | 37 | 16 | 53 |
| 2020 October | 47 | 22 | 69 |
| 2020 September | 23 | 8 | 31 |
| 2020 August | 37 | 20 | 57 |
| 2020 July | 38 | 21 | 59 |
| 2020 June | 31 | 11 | 42 |
| 2020 May | 28 | 12 | 40 |
| 2020 April | 48 | 31 | 79 |
| 2020 March | 29 | 13 | 42 |
| 2020 February | 39 | 24 | 63 |
| 2020 January | 35 | 19 | 54 |
| 2019 December | 44 | 20 | 64 |
| 2019 November | 39 | 37 | 76 |
| 2019 October | 21 | 11 | 32 |
| 2019 September | 32 | 15 | 47 |
| 2019 August | 42 | 22 | 64 |
| 2019 July | 31 | 33 | 64 |
| 2019 June | 19 | 18 | 37 |
| 2019 May | 16 | 11 | 27 |
| 2019 April | 45 | 24 | 69 |
| 2019 March | 26 | 16 | 42 |
| 2019 February | 24 | 14 | 38 |
| 2019 January | 42 | 35 | 77 |
| 2018 December | 23 | 17 | 40 |
| 2018 November | 64 | 31 | 95 |
| 2018 October | 129 | 38 | 167 |
| 2018 September | 36 | 16 | 52 |
| 2018 May | 1 | 0 | 1 |
| 2018 April | 18 | 19 | 37 |
| 2018 March | 36 | 18 | 54 |
| 2018 January | 1 | 0 | 1 |
| 2017 December | 1 | 1 | 2 |
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