Letter to the Editor
Cogan's Syndrome and Spondyloarthritis: Case Report
Síndrome de Cogan y espondiloartritis. Reporte de caso
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"apellidos" => "Pérez Delgado" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S1699258X17302838" "doi" => "10.1016/j.reuma.2017.11.009" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1699258X17302838?idApp=UINPBA00004M" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574319300188?idApp=UINPBA00004M" "url" => "/21735743/0000001500000006/v1_201911250632/S2173574319300188/v1_201911250632/en/main.assets" ] "en" => array:14 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Cogan's Syndrome and Spondyloarthritis: Case Report" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "e170" "paginaFinal" => "e171" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Maria Gabriela Guasamucaro-Castillo, Freddy Alexi Bonito-Torrealba, Teolinda Maldonado, Arnaldo Posadas-Escudero" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Maria Gabriela" "apellidos" => "Guasamucaro-Castillo" "email" => array:1 [ 0 => "gabyguasamucaro@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Freddy Alexi" "apellidos" => "Bonito-Torrealba" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "Teolinda" "apellidos" => "Maldonado" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "Arnaldo" "apellidos" => "Posadas-Escudero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital Central Universitario Dr. Antonio María Pineda, Barquisimeto, Venezuela" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Universidad Centroccidental Lisandro Alvarado, Barquisimeto, Venezuela" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Cogan y espondiloartritis. Reporte de caso" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cogen syndrome (CS) is a systemic inflammatory disease the standard characteristics of which are interstitial keratitis and audiovestibular symptoms similar to those of Ménière's syndrome, with an interval between both manifestations of under 2 years.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">1</span></a> However, atypical forms of the disease exist where ocular or audiovestibular symptoms are different, or the interval between the two is longer than 2 years.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">2</span></a> Its aetiology is unknown, but it has been suggested that there are mechanisms of autoimmunity in the inner ear.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">3</span></a> Apart from steroids, there is no standard treatment alternative for patients with poor response. Cochlear implant offer the opportunity to restore hearing function and communication.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">4</span></a> Association with spondyloarthritis uncommon and we therefore considered it interesting to present this case where satisfactory posterior evolution to the use of steroids, infliximab took place and a cochlear implant was used.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 58-year old man, a cardiologist, with a background of high blood pressure, a tobacco habit and palmoplantar keratoderma since adolescence. He stated he had had inflammatory lumbago and cervical pains since 49 years of age, associated with nonspecific urethritis with no recurring events. At 55 years of age he presented with left keratouveitis. There was evidence of scar-pitting, dactylitis, limitation in flexing hips and a normal breadth of axial movements. Bilateral grade 2 Sacroileitis. Magnetic resonance of the sacroiliac joints: right fatty infiltrate and active left erosion. ESR and RCP rates normal. RA-test and HLA-B27 negative. Anti-HSP-70 positive. Four months after the keratouveitis his hearing was reduced. Audiometry reported moderate hearing loss and bilateral neurosensorial hearing loss. He received 16<span class="elsevierStyleHsp" style=""></span>mg/iv/12<span class="elsevierStyleHsp" style=""></span>h dexametasone for 3 days with improvement. Two weeks later, progressively exacerbated onset of tinnitus. He received treatment with 1<span class="elsevierStyleHsp" style=""></span>g/iv/3 dose of methylprednisolone and later 25<span class="elsevierStyleHsp" style=""></span>mg prednisone daily, 20<span class="elsevierStyleHsp" style=""></span>mg/oral/week methotrexate and 80<span class="elsevierStyleHsp" style=""></span>mg subcut STAT adalimumab and later 40<span class="elsevierStyleHsp" style=""></span>mg/subcut/15 days. He was referred to a hearing clinic but the second audiometric test reported severe bilateral neurosensorial hearing loss, and the third one (3 months later) reported severe right and profound left hearing loss (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>) and as a result the adalimumab was withdrawn. He began treatment with IV cyclophosphamide and took 6 fortnightly doses (4 of 500<span class="elsevierStyleHsp" style=""></span>mg and 2 of 750<span class="elsevierStyleHsp" style=""></span>mg) with no improvement. The patient abandoned treatment due to major depression which was resolved satisfactorily with pharmacological treatment. He was given a cochlear implant a year after the onset of clinical symptoms and this has functioned up until now. Two months later, he started treatment with infliximab (5<span class="elsevierStyleHsp" style=""></span>mg/kg)<span class="elsevierStyleHsp" style=""></span>+<span class="elsevierStyleHsp" style=""></span>20<span class="elsevierStyleHsp" style=""></span>mg/week methotrexate and up to 5<span class="elsevierStyleHsp" style=""></span>mg daily of prednisone was also prescribed. He received treatment with infliximab for 2 years, which was suspended due to the lack of availability of the same, but the patient remained stable and went back to work.</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Cogen syndrome is of low prevalence and is rarely associated with spondyloartritis. Its evolution is generally torpid up to profound hearing loss and there is little response to immunosuppressant treatment. This patient had spondyloartritis with cutaneous involvement (but did not meet with the CASPAR criteria for psoriatic arthritis),<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">5</span></a> and presented with a normal range of axial movements due to the hyperelasticity traits. During the course of the disease he quickly and progressively developed neurosensorial hearing loss which responded to steroid treatment, was associated with anti-HSP70 antibodies and was therefore associated with inner ear autoimmune disease.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">6</span></a> This, together with the keratouveitis, resulted in diagnosis of atypical Cogen syndrome. Few cases regarding this association have been described worldwide and they coincide in the irregular response to immunosuppressant treatment. Post cochlear implant evolution was satisfactory, and has been maintained up until the present day (45 months after its implantation), despite not continuing with the infliximab treatment at present. In this patient, the association between the medical and surgical treatment allowed for the restoration of hearing and the clinical remission of spondyloarthritis, reflecting the importance of multidisciplinary management in complex 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: Guasamucaro-Castillo MG, Bonito-Torrealba FA, Maldonado T, Posadas-Escudero A. Síndrome de Cogan y espondiloartritis. Reporte de caso. Reumatol Clin. 2019;15:e170–e171.</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:2 [ "leyenda" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">RE: right ear; LE: left ear.</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="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col"> \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " colspan="2" align="center" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">1st audiometry</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " colspan="2" align="center" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">2nd audiometry(15 days later)</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " colspan="2" align="center" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">3rd audiometry(3 months later)</th></tr><tr title="table-row"><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black"> \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">RE \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">LE \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">RE \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">LE \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">RE \t\t\t\t\t\t\n \t\t\t\t\t\t</th><th class="td" title="\n \t\t\t\t\ttable-head\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t" scope="col" style="border-bottom: 2px solid black">LE \t\t\t\t\t\t\n \t\t\t\t\t\t</th></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t ; entry_with_role_rowhead " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">Percentage of monoaural loss \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">64 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">34 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">72 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">72 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">79 \t\t\t\t\t\t\n \t\t\t\t</td><td class="td" title="\n \t\t\t\t\ttable-entry\n \t\t\t\t " align="left" valign="\n \t\t\t\t\ttop\n \t\t\t\t">100 \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab2169560.png" ] ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Progression of the Loss of Monoaural Percentage in the Pure Tone and Free Field Audiometry.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0035" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Syndrome of nonsyphilitic interstitial keratitis and vestibuloauditory symptoms" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "D.G. 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Vincenti" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00405-014-3376-9" "Revista" => array:6 [ "tituloSerie" => "Eur Arch Otorhinolaryngol" "fecha" => "2014" "volumen" => "272" "paginaInicial" => "3201" "paginaFinal" => "3207" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25367706" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0055" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Classification criteria for psoriatic arthritis, development of new criteria from a large international study" "autores" => array:1 [ 0 => array:3 [ "colaboracion" => "and the CASPAR Study Group" "etal" => false "autores" => array:6 [ 0 => "W. Taylor" 1 => "D. Gladman" 2 => "P. Helliwell" 3 => "A. Marchesoni" 4 => "P. Mease" 5 => "H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 8 | 4 | 12 |
| 2024 October | 60 | 35 | 95 |
| 2024 September | 65 | 14 | 79 |
| 2024 August | 83 | 26 | 109 |
| 2024 July | 67 | 33 | 100 |
| 2024 June | 80 | 31 | 111 |
| 2024 May | 76 | 25 | 101 |
| 2024 April | 79 | 17 | 96 |
| 2024 March | 57 | 31 | 88 |
| 2024 February | 54 | 32 | 86 |
| 2024 January | 69 | 27 | 96 |
| 2023 December | 49 | 17 | 66 |
| 2023 November | 49 | 24 | 73 |
| 2023 October | 59 | 25 | 84 |
| 2023 September | 82 | 34 | 116 |
| 2023 August | 61 | 20 | 81 |
| 2023 July | 82 | 20 | 102 |
| 2023 June | 65 | 29 | 94 |
| 2023 May | 52 | 18 | 70 |
| 2023 April | 35 | 8 | 43 |
| 2023 March | 60 | 19 | 79 |
| 2023 February | 62 | 23 | 85 |
| 2023 January | 47 | 11 | 58 |
| 2022 December | 64 | 28 | 92 |
| 2022 November | 79 | 25 | 104 |
| 2022 October | 65 | 36 | 101 |
| 2022 September | 53 | 24 | 77 |
| 2022 August | 51 | 57 | 108 |
| 2022 July | 42 | 34 | 76 |
| 2022 June | 40 | 26 | 66 |
| 2022 May | 54 | 36 | 90 |
| 2022 April | 59 | 32 | 91 |
| 2022 March | 60 | 35 | 95 |
| 2022 February | 33 | 24 | 57 |
| 2022 January | 52 | 31 | 83 |
| 2021 December | 45 | 36 | 81 |
| 2021 November | 57 | 37 | 94 |
| 2021 October | 48 | 54 | 102 |
| 2021 September | 48 | 40 | 88 |
| 2021 August | 23 | 32 | 55 |
| 2021 July | 35 | 27 | 62 |
| 2021 June | 42 | 29 | 71 |
| 2021 May | 49 | 50 | 99 |
| 2021 April | 156 | 95 | 251 |
| 2021 March | 84 | 22 | 106 |
| 2021 February | 72 | 13 | 85 |
| 2021 January | 59 | 22 | 81 |
| 2020 December | 62 | 18 | 80 |
| 2020 November | 37 | 11 | 48 |
| 2020 October | 51 | 17 | 68 |
| 2020 September | 116 | 22 | 138 |
| 2020 August | 61 | 23 | 84 |
| 2020 July | 69 | 26 | 95 |
| 2020 June | 24 | 13 | 37 |
| 2020 May | 36 | 18 | 54 |
| 2020 January | 1 | 0 | 1 |
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