Letter to the Editor
Atypical Cogan's Syndrome Associated With Sudden Deafness and Glucocorticoid Response
Síndrome de Cogan atípico asociado a sordera brusca y respuesta a los glucocorticoides
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Cogan in 1945,<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> is a disease of unknown etiology, but probably has an autoimmune basis. It is characterized by the association of non-syphilitic interstitial keratitis and audiovestibular symptoms (similar to Meniere's syndrome), occurring in a period of less than 2 years. It occurs predominantly in young adult Caucasians. There is no predominance of gender.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> When the eye or audiovestibular manifestations are different from what is seen, or the interval between them is more than 2 years, it is called atypic CS.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It may be associated with other systemic manifestations such as aortitis and vasculitis.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> A patient with atypical CS is presented.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient is a 44-year-old male, hypertensive, with axial psoriatic arthritis since age 20 and anterior nongranulomatous uveitis developed in the last 4 years. He attended consultation derived from Otorrhinolaryngology for bilateral Ménière's syndrome resistant to standard therapy, of 3 years of evolution. Audiometry revealed cophosis stood in the left ear and moderate hearing loss in the right ear (RE) at 60<span class="elsevierStyleHsp" style=""></span>dB (the patient wore a hearing aid). Physical examination showed psoriasis and redness in the extremities of both ears, which were rubbery to the touch. No peripheral arthritis was found but he had axial involvement (BASMI: 6). No other data were found upon examination. Laboratory tests showed: ESR of 49<span class="elsevierStyleHsp" style=""></span>mm in the 1st hour and CRP 25<span class="elsevierStyleHsp" style=""></span>mg/L. CBC, serum biochemistry and urinalysis were normal. Autoimmunity: rheumatoid factor, antineutrophil cytoplasmic antibodies, antinuclear antibodies and anti-CCP antibody were all negative. HLA B27 antigen was positive. C3 and C4 were normal. Radiographs of the chest, hands and feet were normal. The sacroiliac X-rays showed stage IV sacroiliitis. Electrocardiogram, echocardiography and computed tomography with intravenous contrast were normal. Atypical CS was diagnosed associated with relapsing polychondritis (no biopsy was performed) and psoriatic arthritis. Methotrexate (MTX) was started orally at doses of 12.5<span class="elsevierStyleHsp" style=""></span>mg/week. At 2 months he presented sudden hearing loss in the RE compared to baseline. The bilateral otoscopy was normal. Audiometry revealed a serious 60<span class="elsevierStyleHsp" style=""></span>dB drop in treble in the RE conversational frequencies. ESR and CRP were 86<span class="elsevierStyleHsp" style=""></span>mm and 45.30<span class="elsevierStyleHsp" style=""></span>mg/L, respectively. Treatment was initiated with boluses of methylprednisolone 1000<span class="elsevierStyleHsp" style=""></span>mg every 24<span class="elsevierStyleHsp" style=""></span>h for 3 days, showing a good response, recovering RE hearing to baseline (determined through a new audiometry) and normalization of acute phase reactants. The patient was discharged with prednisone 30<span class="elsevierStyleHsp" style=""></span>mg/day and tapered the dosage of MTX to 12.5<span class="elsevierStyleHsp" style=""></span>mg/week, remaining asymptomatic after 6 months. Since the first description of CS, more than 220 cases have been described, 92 of them appearing atypically.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Unlike typical CS, the atypical variety is most commonly associated with systemic<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> manifestations and other autoimmune diseases, such as sarcoidosis, rheumatoid arthritis, relapsing polychondritis, juvenile idiopathic arthritis, Sjögren's syndrome and inflammatory bowel disease, among others.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Our case may raise doubts about the diagnosis, given the coexistence of several autoimmune<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,9</span></a> diseases. Psoriatic arthropathy could justify that the patient presented uveitis. Relapsing polychondritis can also present with hearing loss and vertigo, although generally it is a conductive hearing loss and vestibular dysfunction is not as similar to Meniere's. In this patient, the vestibular episodes were intense, with prolonged and bilateral sensorineural hearing loss, preceded by ocular involvement in less than a two year interval, and in the absence of specific complementary data, made us opt for the diagnosis of atypical CS, fulfilling the criteria established by Haynes et al.,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> with 2 associated autoimmune disorders (psoriatic arthritis and relapsing polychondritis) and showing a good response to corticosteroid and immunosuppressive therapy, something relevant given the poor prognosis of deafness.</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: Montes S, Rodríguez-Muguruza S, Soria V, Olivé A. Síndrome de Cogan atípico asociado a sordera brusca y respuesta a los glucocorticoides. Reumatol Clin. 2014;10:267–268.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0005" "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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John H Stone, MD, MPH: UpToDate" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "E. William" 1 => "R. McCallum" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Libro" => array:1 [ "fecha" => "2013" ] ] ] ] ] ] 7 => array:3 [ "identificador" => "bib0040" "etiqueta" => "8" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cogan’ syndrome: an autoinmune inner ear disease" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Greco" 1 => "A. Gallo" 2 => "M. Fusconi" 3 => "G. Magliulo" 4 => "R. Turchetta" 5 => "C. Marinelli" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.autrev.2012.07.012" "Revista" => array:6 [ "tituloSerie" => "Autoimmun Rev" "fecha" => "2013" "volumen" => "12" "paginaInicial" => "396" "paginaFinal" => "400" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22846458" "web" => "Medline" ] ] ] ] ] ] ] ] 8 => array:3 [ "identificador" => "bib0045" "etiqueta" => "9" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Autoimmune sensorineural hearing loss: the otology–rheumatology interface" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "T. Mijovic" 1 => "A. Zeitouni" 2 => "I. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 1 | 4 |
| 2024 October | 56 | 37 | 93 |
| 2024 September | 59 | 16 | 75 |
| 2024 August | 62 | 30 | 92 |
| 2024 July | 72 | 29 | 101 |
| 2024 June | 62 | 32 | 94 |
| 2024 May | 72 | 36 | 108 |
| 2024 April | 57 | 19 | 76 |
| 2024 March | 56 | 34 | 90 |
| 2024 February | 33 | 22 | 55 |
| 2024 January | 48 | 29 | 77 |
| 2023 December | 31 | 28 | 59 |
| 2023 November | 45 | 30 | 75 |
| 2023 October | 72 | 44 | 116 |
| 2023 September | 74 | 47 | 121 |
| 2023 August | 27 | 15 | 42 |
| 2023 July | 54 | 25 | 79 |
| 2023 June | 40 | 23 | 63 |
| 2023 May | 46 | 30 | 76 |
| 2023 April | 29 | 9 | 38 |
| 2023 March | 54 | 37 | 91 |
| 2023 February | 52 | 21 | 73 |
| 2023 January | 33 | 15 | 48 |
| 2022 December | 42 | 45 | 87 |
| 2022 November | 55 | 20 | 75 |
| 2022 October | 46 | 22 | 68 |
| 2022 September | 37 | 22 | 59 |
| 2022 August | 31 | 41 | 72 |
| 2022 July | 31 | 36 | 67 |
| 2022 June | 38 | 31 | 69 |
| 2022 May | 44 | 42 | 86 |
| 2022 April | 52 | 34 | 86 |
| 2022 March | 72 | 41 | 113 |
| 2022 February | 48 | 31 | 79 |
| 2022 January | 52 | 19 | 71 |
| 2021 December | 35 | 43 | 78 |
| 2021 November | 50 | 48 | 98 |
| 2021 October | 63 | 41 | 104 |
| 2021 September | 52 | 45 | 97 |
| 2021 August | 43 | 36 | 79 |
| 2021 July | 36 | 25 | 61 |
| 2021 June | 47 | 44 | 91 |
| 2021 May | 55 | 34 | 89 |
| 2021 April | 101 | 89 | 190 |
| 2021 March | 132 | 23 | 155 |
| 2021 February | 52 | 28 | 80 |
| 2021 January | 44 | 22 | 66 |
| 2020 December | 39 | 14 | 53 |
| 2020 November | 43 | 20 | 63 |
| 2020 October | 22 | 14 | 36 |
| 2020 September | 33 | 17 | 50 |
| 2020 August | 45 | 18 | 63 |
| 2020 July | 29 | 14 | 43 |
| 2020 June | 36 | 19 | 55 |
| 2020 May | 38 | 25 | 63 |
| 2020 April | 29 | 15 | 44 |
| 2020 March | 25 | 6 | 31 |
| 2020 February | 2 | 0 | 2 |
| 2018 May | 4 | 1 | 5 |
| 2018 April | 40 | 11 | 51 |
| 2018 March | 65 | 11 | 76 |
| 2018 February | 19 | 7 | 26 |
| 2018 January | 25 | 3 | 28 |
| 2017 December | 29 | 6 | 35 |
| 2017 November | 34 | 7 | 41 |
| 2017 October | 38 | 8 | 46 |
| 2017 September | 20 | 6 | 26 |
| 2017 August | 38 | 7 | 45 |
| 2017 July | 30 | 13 | 43 |
| 2017 June | 53 | 7 | 60 |
| 2017 May | 52 | 16 | 68 |
| 2017 April | 50 | 6 | 56 |
| 2017 March | 29 | 7 | 36 |
| 2017 February | 24 | 9 | 33 |
| 2017 January | 25 | 14 | 39 |
| 2016 December | 42 | 31 | 73 |
| 2016 November | 37 | 16 | 53 |
| 2016 October | 78 | 23 | 101 |
| 2016 September | 80 | 18 | 98 |
| 2016 August | 62 | 18 | 80 |
| 2016 July | 41 | 26 | 67 |
| 2016 April | 1 | 0 | 1 |
| 2015 December | 3 | 0 | 3 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 20 | 6 | 26 |
| 2015 June | 31 | 29 | 60 |
| 2015 May | 72 | 33 | 105 |
| 2015 April | 34 | 16 | 50 |
| 2015 March | 46 | 18 | 64 |
| 2015 February | 47 | 19 | 66 |
| 2015 January | 40 | 23 | 63 |
| 2014 December | 34 | 23 | 57 |
| 2014 November | 40 | 23 | 63 |
| 2014 October | 56 | 16 | 72 |
| 2014 September | 31 | 22 | 53 |
| 2014 August | 49 | 25 | 74 |
| 2014 July | 49 | 23 | 72 |
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