Letter to the Editor
Libman-Sacks’ endocarditis: A frequently unnoticed complication
Endocarditis de Libman-Sacks: una manifestación frecuentemente inadvertida
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(A) Fast imaging employing steady-state acquisition (FIESTA) sequence in left ventricular (LV) outflow tract, showing a vegetation on the aortic valve (arrow). (B) Short-axis delayed gadolinium-enhanced sequence of the LV, showing an image of subepicardial enhancement at the level of the inferolateral segment suggestive of a vasculitic process (arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Here we describe the case of a 27-year-old female patient with a history of hypertension and cutaneous lupus, who was admitted for a kidney biopsy because she had proteinuria (2081<span class="elsevierStyleHsp" style=""></span>mg/g), microhaematuria (10–20 RBC/field) and mild functional impairment (Cr 1.3<span class="elsevierStyleHsp" style=""></span>mg/dl). The procedure was complicated with a severe perirenal haematoma, which required selective embolization and, subsequently, fever, which was attributed to the procedure and reabsorption of the haematoma; the microbiological tests were negative. The patient developed anaemia (Hb 8<span class="elsevierStyleHsp" style=""></span>g/dl) with normal WBC and platelets. Autoimmunity lab results were: ANA test was positive (1/160); the anti-DNA, positive (33<span class="elsevierStyleHsp" style=""></span>U/ml); and the ENA, SSA, SSB, U1RNP, Sm and SCL70, negative. The lupus anticoagulant (2.3) and the IgG anticardiolipin antibodies (253<span class="elsevierStyleHsp" style=""></span>U/ml) were positive, with decreased C3 levels (70<span class="elsevierStyleHsp" style=""></span>mg/dl). The kidney biopsy showed a type <span class="elsevierStyleSmallCaps">III</span> lupus nephritis. There were only 3 glomeruli (2 with an increased mesangial matrix and one with focal and segmental sclerosis/hyalinosis) with no “wire loop” lesions or glomerular crescent formations. Immunofluorescence showed deposits in the mesangium and the glomerular basement membrane of high intensity for IgG, C3 and C1Q, and mild/moderate intensity for IgA, IgM and C4.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient had cardiac murmur in the auscultation and the fever persisted, so an echocardiogram was performed that showed a bicuspid aortic valve with a vegetation s on the free edge of its 2 leaflets causing a severe insufficiency. Although it was unlikely that this lesion was caused by an infection, owing to the negative result on the blood cultures, a cardiac MRI with contrast dye was performed to clarify the diagnosis. In this examination, in addition to the vegetation there was subepicardial enhancement inferolateral, suggestive of a vasculitic process (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), with no evidence of abscess, pericardial effusion or other complications.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">With a diagnosis of Libman-Sacks endocarditis (LSE), anticoagulant therapy was started, combined with steroids (1<span class="elsevierStyleHsp" style=""></span>mg/kg/day) and mycophenolate (MMF) up to a dose of 1<span class="elsevierStyleHsp" style=""></span>g/12<span class="elsevierStyleHsp" style=""></span>h. After one, she has normal kidney function, the aortic insufficiency has improved to a more moderate gradient.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although the use of steroids in the treatment of systemic lupus erythematosus (SLE) has caused a significant reduction in the incidence of LSE, its current frequency could be up to 10–35% of cases.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,2</span></a> However, its clinical manifestations tend to be mild, and so it often happens unnoticed.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">1</span></a> LSE is usually associated with a long duration of the disease and with the existence of anticardiolipin antibodies (aCL) and antiphospholipid syndrome (APS).<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,3</span></a> The “two-hit hypothesis”, described by Bordin et al.,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">4</span></a> suggests that the pro-thrombotic state generated by aCL (first event) would promote (after a second event such as heart surgery, pregnancy, etc.) the formation of a blood clot on a previously inflamed valvular endothelium.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">3–5</span></a> In the present case, at least from a renal point of view, the disease was not of long duration, but, based on the two-hit hypothesis, one might speculate that, the presence of an aCL-positive during lupus flare-up, plus the damage caused by the haematoma and subsequent embolization, could trigger endocarditis on a valve that is, susceptible because a bicuspid morphology.</p><p id="par0025" class="elsevierStylePara elsevierViewall">This case also illustrates the difficulty of a differential diagnosis vs other valvular malformations or infective endocarditis itself, which may be colonizing on a previous LSE lesion. Although LSE is usually located on the mitral valve, it can also occur on the aortic (as in our case), the tricuspid (less than 10% of cases) and the pulmonary valve.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">1,3</span></a> Because there was no characteristic location or echocardiographic image, the MRI was very useful in clearing up the diagnosis, a finding also documented by other authors.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Regarding treatment, surgery is reserved for cases of severe valvular dysfunction or with large mobile masses with high risk of embolization.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">7</span></a> Although steroid treatment may reduce lesions, it is generally not sufficient for eradicating the embolic risk, so patients should be anticoagulated.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">8</span></a> In our case, and also given the presence of APS and lupus nephritis, treatment with MMF was proposed, with a good renal response and a striking heart response as well, and with a significant improvement of the lesions.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In conclusion, LSE should be included in the differential diagnosis of a patient with SLE and fever, especially if the test is positive for anticardiolipin antibodies and there is a previous triggering factor. In the absence of specific markers, MRI is an important tool for elucidate the diagnosis. Although valvular dysfunction is rare, when it occurs, treatment with steroids and MMF may reduce the lesions and postpone the need for valve replacement. However, given the embolic risk involved, maintaining anticoagulation with close clinical and imaging observation is essential.</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: Gómez-Larrambe N, González-Tabarés L, Millán B, Cobelo C, Armesto V, Pousa M, et al. Endocarditis de Libman-Sacks: una manifestación frecuentemente inadvertida. Nefrologia. 2017;37:217–219.</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" => 1084 "Ancho" => 2399 "Tamanyo" => 141022 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Cardiac MRI with contrast dye. (A) Fast imaging employing steady-state acquisition (FIESTA) sequence in left ventricular (LV) outflow tract, showing a vegetation on the aortic valve (arrow). (B) Short-axis delayed gadolinium-enhanced sequence of the LV, showing an image of subepicardial enhancement at the level of the inferolateral segment suggestive of a vasculitic process (arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0045" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Libman-Sacks endocarditis in systemic lupus erythematosus: prevalence, associations, and evolution" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "I. Moyssakis" 1 => "M.G. Tektonidou" 2 => "V.A. Vassilliou" 3 => "M. Samarkos" 4 => "V. Votteas" 5 => "H.M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 13 | 9 | 22 |
| 2024 October | 114 | 45 | 159 |
| 2024 September | 149 | 30 | 179 |
| 2024 August | 147 | 72 | 219 |
| 2024 July | 101 | 41 | 142 |
| 2024 June | 126 | 43 | 169 |
| 2024 May | 103 | 25 | 128 |
| 2024 April | 79 | 31 | 110 |
| 2024 March | 91 | 26 | 117 |
| 2024 February | 76 | 42 | 118 |
| 2024 January | 85 | 25 | 110 |
| 2023 December | 62 | 34 | 96 |
| 2023 November | 90 | 36 | 126 |
| 2023 October | 140 | 47 | 187 |
| 2023 September | 69 | 27 | 96 |
| 2023 August | 95 | 31 | 126 |
| 2023 July | 155 | 26 | 181 |
| 2023 June | 114 | 25 | 139 |
| 2023 May | 152 | 40 | 192 |
| 2023 April | 79 | 16 | 95 |
| 2023 March | 113 | 21 | 134 |
| 2023 February | 102 | 37 | 139 |
| 2023 January | 109 | 29 | 138 |
| 2022 December | 110 | 28 | 138 |
| 2022 November | 99 | 30 | 129 |
| 2022 October | 106 | 40 | 146 |
| 2022 September | 100 | 44 | 144 |
| 2022 August | 125 | 47 | 172 |
| 2022 July | 137 | 60 | 197 |
| 2022 June | 138 | 46 | 184 |
| 2022 May | 160 | 33 | 193 |
| 2022 April | 215 | 62 | 277 |
| 2022 March | 219 | 58 | 277 |
| 2022 February | 157 | 53 | 210 |
| 2022 January | 162 | 43 | 205 |
| 2021 December | 140 | 35 | 175 |
| 2021 November | 107 | 40 | 147 |
| 2021 October | 117 | 47 | 164 |
| 2021 September | 84 | 40 | 124 |
| 2021 August | 112 | 43 | 155 |
| 2021 July | 128 | 42 | 170 |
| 2021 June | 107 | 33 | 140 |
| 2021 May | 129 | 48 | 177 |
| 2021 April | 276 | 66 | 342 |
| 2021 March | 207 | 29 | 236 |
| 2021 February | 203 | 34 | 237 |
| 2021 January | 121 | 17 | 138 |
| 2020 December | 165 | 20 | 185 |
| 2020 November | 204 | 30 | 234 |
| 2020 October | 138 | 31 | 169 |
| 2020 September | 202 | 24 | 226 |
| 2020 August | 132 | 14 | 146 |
| 2020 July | 193 | 16 | 209 |
| 2020 June | 133 | 15 | 148 |
| 2020 May | 155 | 16 | 171 |
| 2020 April | 317 | 22 | 339 |
| 2020 March | 190 | 22 | 212 |
| 2020 February | 165 | 27 | 192 |
| 2020 January | 206 | 31 | 237 |
| 2019 December | 195 | 30 | 225 |
| 2019 November | 183 | 23 | 206 |
| 2019 October | 140 | 19 | 159 |
| 2019 September | 181 | 19 | 200 |
| 2019 August | 146 | 20 | 166 |
| 2019 July | 123 | 20 | 143 |
| 2019 June | 138 | 24 | 162 |
| 2019 May | 182 | 24 | 206 |
| 2019 April | 205 | 41 | 246 |
| 2019 March | 127 | 33 | 160 |
| 2019 February | 101 | 32 | 133 |
| 2019 January | 94 | 30 | 124 |
| 2018 December | 235 | 44 | 279 |
| 2018 November | 395 | 26 | 421 |
| 2018 October | 398 | 29 | 427 |
| 2018 September | 217 | 22 | 239 |
| 2018 August | 153 | 17 | 170 |
| 2018 July | 122 | 18 | 140 |
| 2018 June | 134 | 18 | 152 |
| 2018 May | 138 | 19 | 157 |
| 2018 April | 178 | 21 | 199 |
| 2018 March | 130 | 16 | 146 |
| 2018 February | 99 | 4 | 103 |
| 2018 January | 89 | 13 | 102 |
| 2017 December | 83 | 6 | 89 |
| 2017 November | 50 | 9 | 59 |
| 2017 October | 59 | 15 | 74 |
| 2017 September | 72 | 13 | 85 |
| 2017 August | 119 | 34 | 153 |
| 2017 July | 87 | 34 | 121 |
| 2017 June | 60 | 29 | 89 |
| 2017 May | 71 | 19 | 90 |
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