Case report
Retinal artery embolization complicating Libman-Sacks endocarditis in a systemic lupus erythematosus patient
Endocardite de Libman-Sacks complicada por embolização da artéria da retina num doente com LES
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No other symptoms were reported, including fever or localized weakness. Fundoscopic examination showed a pale retina with a cherry-red macula (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>) and a diagnosis of right central retinal artery occlusion was made.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Transthoracic and transesophageal echocardiography revealed the presence of a mass attached to the ventricular side of the posterior mitral leaflet, with a vibratory motion, a maximum diameter of 13 mm, irregular shape and heterogeneous echogenicity, consistent with vegetation (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>). The aortic valve had thickened leaflets (<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>) and moderate regurgitation by color Doppler.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Carotid Doppler ultrasound showed no significant atherosclerotic lesions and no other cardioembolic sources were detected.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Inflammatory parameters (white blood cell count 6.9<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/l, C-reactive protein 1.6 mg/dl and erythrocyte sedimentation rate 44 mm/h) were not suggestive of infection. Blood cultures were negative. Autoimmunity study revealed antinuclear antibodies positive at a titer of 1/320, with no other positive antibodies, including negative antiphospholipid antibodies (APA). C3 and C4 levels were normal. Thrombophilia tests including C and S protein levels, antithrombin III and resistance to activated protein C were also normal.</p><p id="par0025" class="elsevierStylePara elsevierViewall">We assumed a diagnosis of Libman-Sacks endocarditis (LSE) and the patient started anticoagulation therapy. A follow-up transesophageal echocardiogram four weeks later showed resolution of the previously described vegetation. There was no recurrence of thromboembolic events.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">SLE is an autoimmune disease that causes multiorgan inflammatory damage. In recent decades, with increasing survival and advances in diagnostic techniques, particularly in echocardiography, cardiac disease associated with SLE has become more evident.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Valvular disease is one of the main cardiac manifestations of SLE and can occur in the form of valvular thickening, masses or noninfective vegetations (LSE), valvular regurgitation and valvular stenosis.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">LSE was first described in 1924 by Libman and Sacks in four patients with SLE and noninfective verrucous vegetations.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Libman-Sacks vegetations develop mainly on the mitral valve, followed by the aortic valve, but may develop on any other valve, on the subvalvular apparatus or on the surface of the endocardium.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> They are usually located on the atrial side of the mitral valve leaflets or the vessel side of the aortic valve leaflets.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">A significant proportion of patients with SLE have LSE detected in autopsy studies (30–50%). However, the real prevalence of LSE remains unknown since most patients with Libman-Sacks vegetations have asymptomatic valve abnormalities.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Moyssakis et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> studied 342 patients with SLE by echocardiography over four years and found an 11% incidence of LSE and an association with lupus duration, disease activity, presence of anticardiolipin antibody and manifestations of antiphospholipid syndrome. Roldan et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> studied 69 patients with SLE by transesophageal echocardiography and found a 43% incidence of LSE, which may be related to the greater accuracy of this modality.</p><p id="par0050" class="elsevierStylePara elsevierViewall">It has been proposed that LSE is due to the formation of fibrin-platelet thrombi on the injured valve, followed by tissue organization and leading to valvular fibrosis, distortion and subsequent dysfunction. Recent studies have shown deposition of immunoglobulins and complement in the valvular structure which subsequently developed LSE and valvular thickening.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The association of LSE and APA has been widely investigated and has been reported in several studies,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4,5</span></a> although others have found no connection.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The role of APA in the pathogenesis of valvular disease is thought to be by promoting thrombus formation on injured valve endothelium and inflammatory changes, rather than a more direct pathogenic role.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Further, the observation that there is a significantly higher prevalence of valvular lesions in patients with antiphospholipid syndrome (APS) secondary to SLE than in those with primary APS may mean that there are SLE-related factors that promote endocardial damage and contribute to this difference.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4</span></a> APA were not detected in our patient, which is in agreement with this theory.</p><p id="par0060" class="elsevierStylePara elsevierViewall">One recognized complication of LSE is the development of secondary infectious endocarditis<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> which increases the complexity of differential diagnosis in a patient with SLE who presents with a valve mass. Infective endocarditis lesions are usually located at the leaflet's line of closure, are homogeneous in echogenicity and may show a vibratory or rotatory motion.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In contrast, LSE lesions are usually located at the base, middle or tip of the leaflets and are variable in shape and size and heterogeneous in echogenicity.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Thus it is imperative to differentiate between these two clinical identities, since management and treatment are quite different. Our patient had no fever, her leukocyte count was normal and blood cultures were negative, which enabled us to reach a diagnosis of LSE and to initiate anticoagulation therapy. Follow-up transesophageal echocardiography provided an assessment of disease progression, revealing the regression of the previously observed vegetation.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The main clinical impact of LSE is related to the probability of lesion progression to valvular dysfunction and the tendency to thromboembolic events, especially stroke or transient ischemic attack.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,4</span></a> The incidence of thromboembolic cerebrovascular events in patients with LSE has been reported as 10–20%<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3</span></a> and a cardioembolic origin was assumed in most cases. In our search of the literature we found several case reports of distal embolization from LSE, the majority reporting cerebral embolization, but none with retinal embolization.</p><p id="par0070" class="elsevierStylePara elsevierViewall">In the present case, there was an occlusion of the right central retinal artery, which originates from the ophthalmic artery, the first intracranial branch of the internal carotid artery. Given the temporal relationship between the onset of right blindness and the echocardiographic finding of a mass adhering to the mitral valve, it was assumed that the retinal artery occlusion was cardioembolic in origin. The patient was started on anticoagulation therapy for secondary thromboprophylaxis and remission of the previously detected vegetation was achieved. To date she has had no new thromboembolic events.</p><p id="par0075" class="elsevierStylePara elsevierViewall">With this case report we highlight the importance of awareness of this entity, allowing rapid referral for cardiovascular examination and thus enabling early diagnosis and appropriate intervention.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Given that most patients with SLE and valvular disease have no cardiac symptoms, a careful cardiovascular examination should be made periodically. Since strokes in patients with SLE are frequent and, on the other hand, valvular thickening and vegetations are common and can act as substrates for cardioembolism, prophylactic therapy with anticoagulation may be an appropriate approach to these patients.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Ethical disclosures</span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Protection of human and animal subjects</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Confidentiality of data</span><p id="par0090" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Right to privacy and informed consent</span><p id="par0095" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article.</p></span></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflicts of interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:2 [ "identificador" => "xres251200" "titulo" => "Abstract" ] 1 => array:2 [ "identificador" => "xpalclavsec238796" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "xres251199" "titulo" => "Resumo" ] 3 => array:2 [ "identificador" => "xpalclavsec238797" "titulo" => "Palavras-chave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 6 => array:3 [ "identificador" => "sec0015" "titulo" => "Ethical disclosures" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0020" "titulo" => "Protection of human and animal subjects" ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Confidentiality of data" ] 2 => array:2 [ "identificador" => "sec0030" "titulo" => "Right to privacy and informed consent" ] ] ] 7 => array:2 [ "identificador" => "sec0035" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2012-10-28" "fechaAceptado" => "2012-11-01" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec238796" "palabras" => array:4 [ 0 => "Systemic lupus erythematosus" 1 => "Libman-Sacks endocarditis" 2 => "Embolization" 3 => "Retinal artery occlusion" ] ] ] "pt" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palavras-chave" "identificador" => "xpalclavsec238797" "palabras" => array:4 [ 0 => "Lúpus eritematoso sistémico" 1 => "Endocardite de Libman-Sacks" 2 => "Embolização" 3 => "Oclusão da artéria da retina" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Libman-Sacks endocarditis (LSE) is the most characteristic cardiac manifestation of systemic lupus erythematosus (SLE). It is usually clinically silent but heart failure due to valvular dysfunction, secondary infective endocarditis and embolic phenomena can complicate valvular abnormalities. We present a patient with SLE and blindness due to right central retinal artery occlusion. Echocardiographic examination revealed a verrucous vegetation on the posterior mitral valve leaflet consistent with LSE. Anticoagulation therapy was started. Echocardiographic regression of the vegetation was observed and there has been no recurrence of thromboembolic events to date.</p>" ] "pt" => array:2 [ "titulo" => "Resumo" "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A endocardite de Libman-Sacks é a manifestação cardíaca mais característica do LES. É habitualmente clinicamente silenciosa, mas a insuficiência cardíaca por disfunção valvular, a endocardite infecciosa secundária e os fenómenos embólicos podem complicar as alterações valvulares. Apresentamos um caso clínico de uma doente com LES e amaurose à direita por trombose da artéria central da retina. O ecocardiograma mostrou uma vegetação verrucosa no folheto posterior da válvula mitral, compatível com endocardite de Libman-Sacks. A doente iniciou terapêutica com anticoagulação, verificou-se regressão da vegetação descrita e não teve recorrência de eventos tromboembólicos até à data.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 647 "Ancho" => 850 "Tamanyo" => 87821 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Right eye fundoscopic examination showing a pale retina with a cherry-red macula.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 803 "Ancho" => 2167 "Tamanyo" => 143310 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Transesophageal echocardiogram showing vegetation (Veg.) on the ventricular side of the posterior mitral leaflet (PML) and aortic valve (AoV) with thickened leaflets.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "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" => true "autores" => array:3 [ 0 => "I. 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Santiago" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s10067-010-1662-5" "Revista" => array:6 [ "tituloSerie" => "Clin Rheumatol" "fecha" => "2011" "volumen" => "30" "paginaInicial" => "165" "paginaFinal" => "171" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/21207090" "web" => "Medline" ] ] ] ] ] ] ] ] 5 => array:3 [ "identificador" => "bib0030" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Systemic lupus erythematosus valve disease by transesophageal echocardiography and the role of antiphospholipid antibodies" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:3 [ 0 => "C.A. Roldan" 1 => "B.K. Shively" 2 => "C.C. 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| Ano/Mês | Html | Total | |
|---|---|---|---|
| 2024 Novembro | 16 | 6 | 22 |
| 2024 Outubro | 69 | 42 | 111 |
| 2024 Setembro | 101 | 35 | 136 |
| 2024 Agosto | 89 | 35 | 124 |
| 2024 Julho | 63 | 34 | 97 |
| 2024 Junho | 52 | 24 | 76 |
| 2024 Maio | 69 | 19 | 88 |
| 2024 Abril | 69 | 35 | 104 |
| 2024 Maro | 78 | 26 | 104 |
| 2024 Fevereiro | 82 | 36 | 118 |
| 2024 Janeiro | 62 | 37 | 99 |
| 2023 Dezembro | 53 | 25 | 78 |
| 2023 Novembro | 62 | 25 | 87 |
| 2023 Outubro | 50 | 19 | 69 |
| 2023 Setembro | 63 | 25 | 88 |
| 2023 Agosto | 60 | 23 | 83 |
| 2023 Julho | 84 | 16 | 100 |
| 2023 Junho | 52 | 14 | 66 |
| 2023 Maio | 73 | 25 | 98 |
| 2023 Abril | 66 | 6 | 72 |
| 2023 Maro | 86 | 30 | 116 |
| 2023 Fevereiro | 54 | 27 | 81 |
| 2023 Janeiro | 55 | 26 | 81 |
| 2022 Dezembro | 97 | 24 | 121 |
| 2022 Novembro | 84 | 25 | 109 |
| 2022 Outubro | 112 | 21 | 133 |
| 2022 Setembro | 109 | 45 | 154 |
| 2022 Agosto | 87 | 26 | 113 |
| 2022 Julho | 70 | 44 | 114 |
| 2022 Junho | 49 | 29 | 78 |
| 2022 Maio | 60 | 37 | 97 |
| 2022 Abril | 73 | 21 | 94 |
| 2022 Maro | 50 | 38 | 88 |
| 2022 Fevereiro | 52 | 22 | 74 |
| 2022 Janeiro | 52 | 23 | 75 |
| 2021 Dezembro | 50 | 35 | 85 |
| 2021 Novembro | 65 | 39 | 104 |
| 2021 Outubro | 57 | 47 | 104 |
| 2021 Setembro | 52 | 35 | 87 |
| 2021 Agosto | 54 | 28 | 82 |
| 2021 Julho | 82 | 27 | 109 |
| 2021 Junho | 54 | 19 | 73 |
| 2021 Maio | 57 | 29 | 86 |
| 2021 Abril | 124 | 38 | 162 |
| 2021 Maro | 114 | 22 | 136 |
| 2021 Fevereiro | 101 | 18 | 119 |
| 2021 Janeiro | 67 | 14 | 81 |
| 2020 Dezembro | 42 | 6 | 48 |
| 2020 Novembro | 58 | 19 | 77 |
| 2020 Outubro | 56 | 24 | 80 |
| 2020 Setembro | 90 | 17 | 107 |
| 2020 Agosto | 53 | 8 | 61 |
| 2020 Julho | 72 | 17 | 89 |
| 2020 Junho | 79 | 16 | 95 |
| 2020 Maio | 104 | 10 | 114 |
| 2020 Abril | 57 | 8 | 65 |
| 2020 Maro | 66 | 11 | 77 |
| 2020 Fevereiro | 193 | 30 | 223 |
| 2020 Janeiro | 80 | 7 | 87 |
| 2019 Dezembro | 81 | 12 | 93 |
| 2019 Novembro | 97 | 8 | 105 |
| 2019 Outubro | 118 | 8 | 126 |
| 2019 Setembro | 147 | 12 | 159 |
| 2019 Agosto | 77 | 4 | 81 |
| 2019 Julho | 100 | 14 | 114 |
| 2019 Junho | 94 | 10 | 104 |
| 2019 Maio | 130 | 12 | 142 |
| 2019 Abril | 89 | 19 | 108 |
| 2019 Maro | 162 | 14 | 176 |
| 2019 Fevereiro | 159 | 15 | 174 |
| 2019 Janeiro | 107 | 6 | 113 |
| 2018 Dezembro | 119 | 10 | 129 |
| 2018 Novembro | 127 | 10 | 137 |
| 2018 Outubro | 156 | 23 | 179 |
| 2018 Setembro | 48 | 12 | 60 |
| 2018 Agosto | 51 | 10 | 61 |
| 2018 Julho | 52 | 6 | 58 |
| 2018 Junho | 77 | 7 | 84 |
| 2018 Maio | 79 | 9 | 88 |
| 2018 Abril | 136 | 19 | 155 |
| 2018 Maro | 84 | 10 | 94 |
| 2018 Fevereiro | 58 | 4 | 62 |
| 2018 Janeiro | 54 | 5 | 59 |
| 2017 Dezembro | 106 | 13 | 119 |
| 2017 Novembro | 55 | 11 | 66 |
| 2017 Outubro | 28 | 9 | 37 |
| 2017 Setembro | 58 | 7 | 65 |
| 2017 Agosto | 64 | 12 | 76 |
| 2017 Julho | 36 | 6 | 42 |
| 2017 Junho | 55 | 16 | 71 |
| 2017 Maio | 76 | 10 | 86 |
| 2017 Abril | 53 | 2 | 55 |
| 2017 Maro | 69 | 15 | 84 |
| 2017 Fevereiro | 107 | 10 | 117 |
| 2017 Janeiro | 47 | 9 | 56 |
| 2016 Dezembro | 39 | 17 | 56 |
| 2016 Novembro | 43 | 23 | 66 |
| 2016 Outubro | 57 | 14 | 71 |
| 2016 Setembro | 99 | 9 | 108 |
| 2016 Agosto | 44 | 6 | 50 |
| 2016 Julho | 13 | 6 | 19 |
| 2016 Junho | 5 | 5 | 10 |
| 2016 Maio | 18 | 0 | 18 |
| 2016 Abril | 50 | 4 | 54 |
| 2016 Maro | 97 | 10 | 107 |
| 2016 Fevereiro | 127 | 21 | 148 |
| 2016 Janeiro | 93 | 12 | 105 |
| 2015 Dezembro | 105 | 12 | 117 |
| 2015 Novembro | 98 | 5 | 103 |
| 2015 Outubro | 116 | 12 | 128 |
| 2015 Setembro | 120 | 13 | 133 |
| 2015 Agosto | 115 | 15 | 130 |
| 2015 Julho | 144 | 6 | 150 |
| 2015 Junho | 89 | 2 | 91 |
| 2015 Maio | 76 | 8 | 84 |
| 2015 Abril | 87 | 7 | 94 |
| 2015 Maro | 87 | 11 | 98 |
| 2015 Fevereiro | 129 | 9 | 138 |
| 2015 Janeiro | 90 | 12 | 102 |
| 2014 Dezembro | 80 | 11 | 91 |
| 2014 Novembro | 78 | 8 | 86 |
| 2014 Outubro | 91 | 17 | 108 |
| 2014 Setembro | 76 | 16 | 92 |
| 2014 Agosto | 71 | 13 | 84 |
| 2014 Julho | 59 | 16 | 75 |
| 2014 Junho | 62 | 16 | 78 |
| 2014 Maio | 58 | 17 | 75 |
| 2014 Abril | 67 | 11 | 78 |
| 2014 Maro | 144 | 15 | 159 |
| 2014 Fevereiro | 127 | 10 | 137 |
| 2014 Janeiro | 123 | 15 | 138 |
| 2013 Dezembro | 126 | 13 | 139 |
| 2013 Novembro | 124 | 12 | 136 |
| 2013 Outubro | 103 | 9 | 112 |
| 2013 Setembro | 107 | 19 | 126 |
| 2013 Agosto | 108 | 21 | 129 |
| 2013 Julho | 94 | 22 | 116 |
| 2013 Junho | 73 | 21 | 94 |
| 2013 Maio | 109 | 38 | 147 |
| 2013 Abril | 32 | 15 | 47 |
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