Letter to the Editor
Bilateral Sudden Sensorineural Hearing Loss in a Patient With Microangiopathic Antiphospholipid Syndrome
Anacusia súbita neurosensorial bilateral en un paciente con síndrome antifosfolípido primario microangiopático
a Departamento de Reumatología, Hospital General de Zona N.° 2-A «Troncoso», IMSS, Mexico City, Mexico
b Departamento de Medicina Interna, Hospital de Especialidades, Centro Médico Nacional Siglo XXI, IMSS, Mexico City, Mexico
c Coordinación de Planeación de Infraestructura Médica, Instituto Mexicano del Seguro Social, Mexico City, Mexico
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"etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Anacusia súbita neurosensorial bilateral en un paciente con síndrome antifosfolípido primario microangiopático" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1800 "Ancho" => 1800 "Tamanyo" => 294406 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) T1-weighted magnetic resonance imaging sequence showing a hyperintense area of microvascular damage in white matter of bilateral frontal and parietal lobes; (b) inversion recovery-weighted magnetic resonance imaging sequence showing lesions in right temporal lobe; (c) magnetic resonance angiography showing anterior cerebral circulation with no large vessel involvement; (d) magnetic resonance angiography showing normal circle of Willis.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Sudden sensorineural hearing loss (SSHL) is defined as a sudden deterioration in auditory function of at least 30<span class="elsevierStyleHsp" style=""></span>dB; bilateral presentation (BSSHL) is extremely rare (<5% of all cases of SSHL).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> The known etiological agents are viral infections, tympanic membrane perforation, vascular disorders and autoimmune diseases.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The autoimmune etiology was proposed by Ernst Lehnhardt in a patient with unilateral SSHL, who subsequently developed the same condition in the other ear.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> He postulated that the damage to the first ear led to the development of antibodies that affected the contralateral ear. The first associations of BSSHL with rheumatic diseases were reported in the 1980s in patients with systemic lupus erythematosus (SLE) and anticardiolipin antibodies (aCL).<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> It was during the same period of time that Hughes pointed out the unusual presentations of thromboses, miscarriages and lupus anticoagulant in patients with SLE, an observation that would lay the foundations for the characterization of antiphospholipid syndrome (APS). Since then, the concept of APS has evolved considerably. In recent years, a variant with exclusively microangiopathic involvement (MAPS), in which virtually any organ can be affected, has been recognized.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 54-year-old man presented with BSSHL, headache and vertigo. He had had acute pancreatitis with secondary diabetes mellitus 4 years earlier and had a 7-year history of primary hypothyroidism. He underwent a neurotologic examination and, following audiometry, was diagnosed with bilateral neurosensory hearing loss, which was treated with transtympanic dexamethasone. A specialist in infectious diseases prescribed ganciclovir at a dose of 12<span class="elsevierStyleHsp" style=""></span>mg/kg/day for 10 days<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>prednisone at 50<span class="elsevierStyleHsp" style=""></span>mg/day for 4 weeks. Nerve conduction studies revealed short latency auditory evoked potentials with no response. Speech audiometry showed an absence of response to maximum intensities. The patient was referred to lip and face-reading therapy and to rheumatology because he had been found to have aCL. In the latter department, we reached a diagnosis of APS on the basis of high titers of IgM anti-β2 glycoprotein and aCL, a positive test for lupus anticoagulant and a prolonged activated partial thromboplastin time (aPTT). After immunological studies (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>), we ruled out the presence of lupus. In addition, in magnetic resonance imaging (MRI), we observed subcortical hyperintensity in bilateral frontal and parietal lobes, with normal vascular behavior and enhancement, findings that demonstrate microvascular involvement (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). As the patient had never had large vessel thrombosis, we concluded that what he had was primary APS in its microangiopathic variant.</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">We report a representative case of MAPS, diagnosed on the basis of the high titers of IgM anti-β2 glycoprotein and aCL, the presence of lupus anticoagulant, prolonged aPTT and microvascular signs. Central nervous system involvement was made evident by the typical microvascular changes on MRI, characterized by lesions predominantly in the white matter because of the greater vulnerability of that region to ischemia.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Bilateral SSHL is a very rare finding and, although its association with aCL is well established, the pathogenic mechanisms remain a mystery. It has been suggested that antiphospholipid antibodies (aPL) activate the endothelium of the cochlear blood vessels and that this overregulation produces local microthrombi and ischemia in the inner ear.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> With respect to the pancreatic manifestations, once other nosological entities have been ruled out, it reasonable to consider that they may be related to MAPS. In this respect, the first case of pancreatic compromise in APS was described by Bird et al.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> and, since then, several cases have been reported. Autopsies show chronic inflammation and thrombosis in pancreatic arteries in the absence of vasculitis.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Concerning the presence of hypothyroidism, the clinical significance of aPL in thyroid autoimmune disease is still a subject of controversy. Current evidence suggests that the production of anti-thyroglobulin and anti-microsomal antibodies is accompanied by aPL synthesis as an epiphenomenon.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In conclusion, BSSHL is a medical emergency with an ominous prognosis. There is no consensus with respect to the treatment, but it is highly recommendable that anticoagulation therapy begin immediately. Immunosuppressive therapy has not been found to be useful in the reported cases.</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: Galicia-López A, Anda-Garay JC, García de la Peña M. Anacusia súbita neurosensorial bilateral en un paciente con síndrome antifosfolípido primario microangiopático. Reumatol Clin. 2016;12:175–177.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1800 "Ancho" => 1800 "Tamanyo" => 294406 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) T1-weighted magnetic resonance imaging sequence showing a hyperintense area of microvascular damage in white matter of bilateral frontal and parietal lobes; (b) inversion recovery-weighted magnetic resonance imaging sequence showing lesions in right temporal lobe; (c) magnetic resonance angiography showing anterior cerebral circulation with no large vessel involvement; (d) magnetic resonance angiography showing normal circle of Willis.</p>" ] ] 1 => 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:2 [ "leyenda" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">GPL, IgG phospholipid units; MPL, IgM phospholipid units.</p>" "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 " align="left" valign="top" scope="col" style="border-bottom: 2px solid black">Test \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">Result \t\t\t\t\t\t\n \t\t\t\t</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Antinuclear antibodies \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">80 homogeneous, 40 cytoplasmic \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">Anti-DNA antibodies \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Negative \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">Anti-Sm antibodies \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">Negative \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">Lupus anticoagulant \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">1.69 (positive <1.2) \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">IgM anti-β2 glycoprotein \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">3350 (<8<span class="elsevierStyleHsp" style=""></span>U/mL) \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">IgG anti-β2 glycoprotein \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">6.8 (14.3<span class="elsevierStyleHsp" style=""></span>U/mL) \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">IgM anticardiolipin \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">>255<span class="elsevierStyleHsp" style=""></span>MPL (positive >15) \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">IgG anticardiolipin \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">46.3<span class="elsevierStyleHsp" style=""></span>GPL (positive >20.1) \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">Complement C3 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">296.7<span class="elsevierStyleHsp" style=""></span>mg/dL (90–180) \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">Complement C4 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="table-entry " align="left" valign="top">29.8<span class="elsevierStyleHsp" style=""></span>mg/dL (10–40) \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab1040762.png" ] ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Immunological Studies.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Bilateral sudden sensorineural hearing loss: review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "S.A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 16 | 19 | 35 |
| 2024 September | 42 | 29 | 71 |
| 2024 August | 61 | 51 | 112 |
| 2024 July | 55 | 39 | 94 |
| 2024 June | 67 | 51 | 118 |
| 2024 May | 56 | 42 | 98 |
| 2024 April | 57 | 44 | 101 |
| 2024 March | 45 | 43 | 88 |
| 2024 February | 51 | 34 | 85 |
| 2024 January | 36 | 33 | 69 |
| 2023 December | 44 | 51 | 95 |
| 2023 November | 42 | 47 | 89 |
| 2023 October | 46 | 41 | 87 |
| 2023 September | 61 | 62 | 123 |
| 2023 August | 30 | 38 | 68 |
| 2023 July | 36 | 39 | 75 |
| 2023 June | 46 | 40 | 86 |
| 2023 May | 47 | 46 | 93 |
| 2023 April | 30 | 20 | 50 |
| 2023 March | 49 | 44 | 93 |
| 2023 February | 52 | 37 | 89 |
| 2023 January | 33 | 35 | 68 |
| 2022 December | 44 | 54 | 98 |
| 2022 November | 84 | 39 | 123 |
| 2022 October | 48 | 43 | 91 |
| 2022 September | 46 | 30 | 76 |
| 2022 August | 46 | 47 | 93 |
| 2022 July | 47 | 53 | 100 |
| 2022 June | 53 | 50 | 103 |
| 2022 May | 40 | 43 | 83 |
| 2022 April | 42 | 59 | 101 |
| 2022 March | 82 | 79 | 161 |
| 2022 February | 45 | 36 | 81 |
| 2022 January | 53 | 29 | 82 |
| 2021 December | 45 | 34 | 79 |
| 2021 November | 42 | 48 | 90 |
| 2021 October | 66 | 53 | 119 |
| 2021 September | 39 | 25 | 64 |
| 2021 August | 60 | 42 | 102 |
| 2021 July | 45 | 30 | 75 |
| 2021 June | 45 | 31 | 76 |
| 2021 May | 66 | 49 | 115 |
| 2021 April | 142 | 92 | 234 |
| 2021 March | 87 | 46 | 133 |
| 2021 February | 55 | 29 | 84 |
| 2021 January | 57 | 23 | 80 |
| 2020 December | 66 | 18 | 84 |
| 2020 November | 56 | 22 | 78 |
| 2020 October | 60 | 22 | 82 |
| 2020 September | 72 | 32 | 104 |
| 2020 August | 38 | 21 | 59 |
| 2020 July | 30 | 16 | 46 |
| 2020 June | 35 | 15 | 50 |
| 2020 May | 38 | 23 | 61 |
| 2020 April | 30 | 17 | 47 |
| 2020 March | 31 | 6 | 37 |
| 2020 February | 1 | 0 | 1 |
| 2018 May | 8 | 0 | 8 |
| 2018 April | 35 | 13 | 48 |
| 2018 March | 108 | 8 | 116 |
| 2018 February | 27 | 6 | 33 |
| 2018 January | 25 | 5 | 30 |
| 2017 December | 27 | 5 | 32 |
| 2017 November | 25 | 9 | 34 |
| 2017 October | 26 | 7 | 33 |
| 2017 September | 28 | 8 | 36 |
| 2017 August | 23 | 7 | 30 |
| 2017 July | 31 | 18 | 49 |
| 2017 June | 50 | 27 | 77 |
| 2017 May | 49 | 15 | 64 |
| 2017 April | 45 | 29 | 74 |
| 2017 March | 43 | 15 | 58 |
| 2017 February | 18 | 15 | 33 |
| 2017 January | 33 | 5 | 38 |
| 2016 December | 63 | 13 | 76 |
| 2016 November | 52 | 19 | 71 |
| 2016 October | 51 | 9 | 60 |
| 2016 September | 53 | 9 | 62 |
| 2016 August | 38 | 4 | 42 |
| 2016 July | 26 | 14 | 40 |
| 2016 June | 1 | 0 | 1 |
| 2016 May | 1 | 0 | 1 |
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