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Retinal arterial and venous obstruction as the first manifestation of an antiphospholipid antibody syndrome
Obstrucción arterial y venosa retiniana como primera manifestación de un síndrome de anticuerpos antifosfolípidos
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T1 post-contrast MRI shows inflammatory enhancement of orbital tissues (asterisk), perineural of the optic nerve (small arrow) and of the left orbital apex (large arrow) (A) and of the left cavernous sinus (B) (white arrow).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "C. Ruiz-Arranz, J.A. Reche-Sainz, M.C. de Uña-Iglesias, A. Ortueta-Olartecoechea, A. Muñoz-Gallego, M. Ferro-Osuna" "autores" => array:6 [ 0 => array:2 [ "nombre" => "C." "apellidos" => "Ruiz-Arranz" ] 1 => array:2 [ "nombre" => "J.A." "apellidos" => "Reche-Sainz" ] 2 => array:2 [ "nombre" => "M.C." "apellidos" => "de Uña-Iglesias" ] 3 => array:2 [ "nombre" => "A." "apellidos" => "Ortueta-Olartecoechea" ] 4 => array:2 [ "nombre" => "A." "apellidos" => "Muñoz-Gallego" ] 5 => array:2 [ "nombre" => "M." 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There is a diffuse papilla elevation with posterior vitritis. In part B, B-mode ultrasound shows optic nerve elevation without posterior acoustic shadowing, ruling out calcifications.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "F.J. Valentín-Bravo, C. García-Álvarez, M. Para-Prieto, M.A. Sánchez-Ronco, M.A. Saornil-Álvarez" "autores" => array:5 [ 0 => array:2 [ "nombre" => "F.J." "apellidos" => "Valentín-Bravo" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "García-Álvarez" ] 2 => array:2 [ "nombre" => "M." 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Pérez-Crespo, C. Gutiérrez-Ortiz" "autores" => array:2 [ 0 => array:3 [ "nombre" => "A." "apellidos" => "Pérez-Crespo" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "C." "apellidos" => "Gutiérrez-Ortiz" "email" => array:1 [ 0 => "consuelogutierre@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Licenciada en Medicina y Cirugía. Hospital Príncipe de Asturias, Servicio de Oftalmología, Alcalá de Henares, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Doctora en Medicina y Cirugía. Hospital Príncipe de Asturias, Servicio de Oftalmología, Alcalá de Henares, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Obstrucción arterial y venosa retiniana como primera manifestación de un síndrome de anticuerpos antifosfolípidos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 426 "Ancho" => 750 "Tamanyo" => 31203 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Image of the fundus of the right eye at 12 months of follow-up.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">The antiphospholipid syndrome (APS) was defined by Hughes<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> in 1983 as a form of autoantibody-induced thrombophilia clinically characterized by thrombosis and recurrent obstetric complications. The antibodies involved in its pathogenesis are the so-called antiphospholipids (APS), directed against the phospholipids of cell membranes. Several types of APS are known and are detected by three analytical tests: anti-cardiolipin antibodies (ACL), anti-glycoprotein (ß2) and lupus anticoagulant (LA).<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> APS can be primary or secondary,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> depending on whether it occurs in isolation or in association with other autoimmune diseases.</p><p id="par0010" class="elsevierStylePara elsevierViewall">APS can cause arterial or venous thrombosis in any organ system, and even affect heart valves.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> The most frequent venous event is deep vein thrombosis of the lower limbs, while the most common arterial event is stroke. Ocular pathology is variable, and may be asymptomatic or produce blurred vision, episodes of amaurosis fugax or campimetric defects. The posterior segment is the most affected, classically associating the occlusion of the vein or central artery of the retina or one of its branches. It can also cause an optic neuropathy.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The clinic case of a female who exhibited a mixed vascular obstructive picture of cilioretinal artery and venous component as the first manifestation of an antiphospholipid syndrome is presented.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Clinic case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">Female, 34, with unremarkable personal history or previous abortion, consulted for sudden and painless decrease in visual acuity (VA) of the right eye (RE); she also reported a similar self-limited episode two weeks earlier. Examination showed a VA in the RE of finger count at 1 m and of the unit in the left eye (LE) and afferent pupil defect in the RE. No alteration was found in the anterior pole. The ocular fundus (OF) of the RE showed hemorrhages over the upper and lower temporal arches, venous tortuosity (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) and retinal pallor in the area irrigated by the cilioretinal artery and macular "cherry red spot". The OF of the LE was normal. Fluorescein angiography (FAG) showed the venous obstruction of the LE and a blockage of the cilioretinal artery (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In the visual field (VF) a defect appeared in the region corresponding to the RE papillomacular bundle, with the LE bundle being normal (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The optical coherence tomography showed slight disc edema in the upper and lower quadrants of the RE and a normal macular profile.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">In view of the patient's signs and symptoms, a complete analysis, reaginic, immunological and coagulation tests were requested, as well as consultations with the Hematology and Cardiology Departments. The latter ruled out thromboembolic phenomena caused by arrhythmias, carotid stenosis or other cardiac pathologies.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Hematology requested the following tests to rule out congenital or acquired states of hypercoagulability: activated protein C resistance, factor V Leiden and prothrombin gene variant 20210A, protein C activity, free protein S concentration, AT activity, FVIII activity, lupus anticoagulant, anti-cardiolipin antibodies and β2-glycoprotein I antibodies (IgG and IgM) and plasma homocysteine concentration. ACL antibodies (ACL IgG > 20) and AL antibodies were positive. The determination of the AP (ACL and AL) was positive in a second test three months after the first test.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The patient was diagnosed with antiphospholipid antibody syndrome because she presented a mixed retinal, arterial and venous thrombosis, and because the antibodies characteristic of the syndrome were positive. No other autoimmune diseases were found to be associated.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Antiaggregate treatment (acetylsalicylic acid, 100 mg/d), anticoagulant (low molecular weight heparin at therapeutic doses and acenocoumarol 2 mg/d) and corticoid (oral prednisone, 50 mg/d) were initiated, obtaining a good therapeutic response. During the follow-up, which was 12 months for our part, antiaggregation and corticoids were withdrawn, and anticoagulation was maintained by adjusting the dose to achieve an INR between 2.5 and 3.5. final VA was of the unit for both eyes, with a restoration to normal of the OF image of the RE (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">APS is an autoimmune entity with defined clinical characteristics associated with measurable antibodies. It is diagnosed using the Sydney (2006)<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> classification criteria, which require at least one clinical criterion (thrombosis, obstetric complications) and one analytical criterion (lupus anticoagulant, anti-cardiolipin antibodies, ß2-Glycoprotein antibodies; determined on two or more occasions at least 12 weeks apart). The management of these patients should be individualized, since in addition to the APs, other risk factors for thrombosis are usually taken into account, such as the presence of an underlying autoimmune disease and cardiovascular risk factors, eliminating the so-called modifiable factors to reduce the risk of thrombosis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Its approach is twofold, primary prophylaxis in patients who have not had previous thrombosis, and secondary treatment or prophylaxis in patients with previous thrombosis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Management is based on anticoagulation (low-molecular-weight heparin plus warfarin or acenocoumarol) with or without platelet antiaggregation (low-dose acetylsalicylic acid or clopidogrel), but in some patients immunoglobulins, corticoids, hydroxychloroquine or anticoagulants that inhibit factor Xa are administered. Direct-acting anticoagulants are not recommended.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> In the case described, the ophthalmological alteration was the key to reaching the diagnosis, since the patient had no other systemic clinic, history of previous abortions, or cardiovascular risk factors. On the other hand, the simultaneity of arterial and retinal venous obstruction in this case is unique. Vascular occlusions in young subjects without risk factors should always lead to consider a basic systemic pathology. When the suspicion rate is high, a first inconclusive result should not rule out the entity and the analysis should be repeated.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Accordingly, APS is a disease with a wide range of clinical manifestations that can contribute to increased mortality. Ocular manifestations may be its sign of presentation, as was the case in our patient, so we must bear this in mind in the differential diagnosis of vaso-occlusive retinopathies, especially in young subjects without vascular risk factors<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> and request the relevant analyses and tests which should include a study to rule out cardiac or carotid thrombo-embolic pathology and congenital or acquired states of hypercoagulability. The patient must remain anticoagulated for life and multidisciplinary management is important for better adjustment of treatment, to avoid recurrent thrombotic events and their potentially fatal consequences.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Funding</span><p id="par0055" class="elsevierStylePara elsevierViewall">This work was not funded in any way.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflict of interests</span><p id="par0060" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1541335" "titulo" => "Abstract" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1394322" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1541334" "titulo" => "Resumen" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1394323" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-04-22" "fechaAceptado" => "2020-06-08" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1394322" "palabras" => array:6 [ 0 => "Antiphospholipid syndrome" 1 => "Antiphospholipid antibodies" 2 => "Thrombosis" 3 => "Retinal vessels" 4 => "Antiplatelet therapy" 5 => "Anticoagulation" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1394323" "palabras" => array:6 [ 0 => "Síndrome antifosfolípido" 1 => "Anticuerpos antifosfolípidos" 2 => "Trombosis" 3 => "Vasos retinianos" 4 => "Antiagregación" 5 => "Anticoagulación" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:3 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Case report</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">A 34 year-old woman presented with decrease in visual acuity in her right eye (RE). Her past medical history was unremarkable. Dilated fundus examination revealed a central venous occlusion and an obstruction of the cilioretinal artery. Given the patient age, a cardiology and haematology screen was obtained to rule out hypercoagulation disorders and thromboembolic disease. Antiplatelet, anticoagulant, and corticosteroid therapy were started. The laboratory result was positive for anticardiolipin and antiphospholipid antibody. A diagnosis of antiphospholipid syndrome (APS) was made.</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Discussion</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Antiphospholipid syndrome may cause thrombosis in any organ. The involvement of the retinal vessels may be the first manifestation of this entity. This diagnosis is important to prevent recurrent thrombotic events.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Discussion" ] ] ] "es" => array:3 [ "titulo" => "Resumen" "resumen" => "<span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Caso clínico</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Mujer de 34 años sin antecedentes de interés que acudió por disminución de visión del ojo derecho. El examen reveló una obstrucción de vena central de la retina y de la arteria ciliorretiniana. Dada la edad de la paciente se consultó con cardiología y hematología para descartar un estado de hipercoagulabilidad y patología trombo-embólica. La analítica fue positiva para los anticuerpos anticardiolipina y antifosfolípidos. Se diagnosticó de un síndrome de anticuerpos antifosfolípidos. Se inició tratamiento antiagregante, anticoagulante y corticoideo.</p></span> <span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Discusión</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">El síndrome antifosfolípidos puede causar trombosis en cualquier órgano. La afectación de los vasos retinianos puede ser la primera manifestación. Su diagnóstico es importante para evitar fenómenos trombóticos recurrentes.</p></span>" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "abst0015" "titulo" => "Caso clínico" ] 1 => array:2 [ "identificador" => "abst0020" "titulo" => "Discusión" ] ] ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Pérez-Crespo A, Gutiérrez-Ortiz C. Obstrucción arterial y venosa retiniana como primera manifestación de un síndrome de anticuerpos antifosfolípidos. Arch Soc Esp Oftalmol. 2021;96:380–383.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 442 "Ancho" => 1250 "Tamanyo" => 60839 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Image of the right eye fundus (left): Central retinal vein obstruction, bleeding over the upper and lower temporal arches, venous tortuosity, retinal pallor and "cherry red" macular stain from the blockage of the cilioretinal artery. Fluorescein angiography of the right eye (right): papillophlebitis with obstruction of the cilioretinal artery and venous component.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1346 "Ancho" => 2083 "Tamanyo" => 204107 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Right eye campimetry (left): defect in the region corresponding to the papillomacular bundle.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 426 "Ancho" => 750 "Tamanyo" => 31203 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Image of the fundus of the right eye at 12 months of follow-up.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Thrombosis, abortion, cerebral disease, and the lupus anticoagulant" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "G.R.V. 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