Case and Research Letter
Atypical Ulcerated Lesions in a Patient With Cogan Syndrome
Lesiones ulcerativas atípicas en un paciente con síndrome de Cogan
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Solano-López, A. Montes-Torres, E. Tomero, J. Fraga, D. De Argila, E. Daudèc)n" "autores" => array:6 [ 0 => array:4 [ "nombre" => "G." "apellidos" => "Solano-López" "email" => array:1 [ 0 => "guitje1@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Montes-Torres" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "E." "apellidos" => "Tomero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Fraga" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 4 => array:3 [ "nombre" => "D." "apellidos" => "De Argila" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 5 => array:3 [ "nombre" => "E." "apellidos" => "Daudèc)n" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Department of Dermatology, Hospital Universitario de la Princesa, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Department of Rheumatology, Hospital Universitario de la Princesa, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Department of Pathology, Hospital Universitario de la Princesa, Madrid, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Lesiones ulcerativas atípicas en un paciente con síndrome de Cogan" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 616 "Ancho" => 1600 "Tamanyo" => 270328 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Ulcerated epidermis with a diffuse inflammatory infiltrate in the dermis. (B) Infiltrates formed of neutrophils, histiocytes, lymphocytes, and occasional multinucleated giant cells.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cogan syndrome (CS) is a rare vasculitis characterized by recurrent interstitial keratitis and audiovestibular symptoms including hearing loss, tinnitus, and vertigo. To date, cutaneous lesions have been only rarely described associated with this autoimmune disease.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> We present the case of a patient with CS and multiple ulcerated lesions.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 39-year-old white Spanish woman diagnosed with CS at age 31 years, with stromal keratitis and severe bilateral hearing loss, consulted for a 2-week history of ulcers that had started as pustules on the legs and then spread to the thighs, arms, abdomen, and back. At the time of onset, the patient was on treatment with methotrexate (25<span class="elsevierStyleHsp" style=""></span>mg/wk), prednisone (10<span class="elsevierStyleHsp" style=""></span>mg/d), and tozilizumab (8<span class="elsevierStyleHsp" style=""></span>mg/kg/mo). Physical examination revealed numerous pustules and ulcerated necrotic lesions on the legs, thighs, arms, back, and abdomen (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). On suspicion of ecthyma, topical antibiotics and oral cloxacillin were prescribed. Smear cultures for bacteria, fungi, and mycobacteria were all negative. Additional tests were within normal limits and the chest radiograph was normal. Two weeks later the patient presented further pustules and enlargement of those previously present. At no time did she report fever or malaise. Histopathology of a biopsy from an ulcer on the left thigh revealed an ulcerated epidermis with epidermal necrosis and pustules at the border, with an abscessified area of skin and a diffuse dermal inflammatory infiltrate formed by neutrophils, histiocytes, lymphocytes, and occasional multinucleated giant cells (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A and B). No clear signs of vasculitis were observed. Immunofluorescence was negative. Bacterial cultures from the cutaneous biopsy and new smears were positive for <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span>/<span class="elsevierStyleItalic">Staphylococcus haemolyticus</span>, and <span class="elsevierStyleItalic">Streptococcus pyogenes</span>/<span class="elsevierStyleItalic">Pseudomonas putida fluorescens</span>, respectively. Oral cefuroxime and ciprofloxacin were started but the lesions showed no improvement. It was then that prednisone (at a dose of up to 1<span class="elsevierStyleHsp" style=""></span>mg/kg/d) was prescribed. The patient responded favorably, and in 2 weeks the lesions had completely re-epithelized except for an ulcer in the surgical wound of the biopsy and a linear ulcer on the left arm that had developed after trauma a week earlier, suggesting a pathergy phenomenon (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). At follow-up, no lesions were observed. At the time of writing, the patient remains asymptomatic and continues her usual therapy.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Diagnosis of the cutaneous lesions in our patient was difficult, and ecthyma was our initial provisional diagnosis. The lack of response•and even a deterioration•of the lesions after antibiotic therapy, the pathergy phenomenon, and the favorable response to steroids led us consider a pyoderma gangrenosum (PG)-like neutrophilic dermatosis. However, the multiple lesions and the histopathology were not conclusive for either PG or other forms of neutrophilic dermatosis. Multiple lesions have rarely been described in PG.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> In 70% of cases, PG is associated with an underlying disease such as inflammatory bowel disease (IBD), inflammatory arthritis, or a hematologic malignancy.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3•5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Tirelli et al.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> found about 250 reports of patients with CS, only 13 of whom had concomitant chronic IBD; of these, none experienced improvement after therapy.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of CS are unknown. Initially, the disease was thought to be caused by an infection, but it is now considered to be an autoimmune disorder.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In addition to the ocular and audiovestibular involvement, numerous systemic manifestations have been reported in CS, most commonly of cardiovascular, neurological, or gastrointestinal origin. Approximately 70% of patients have an underlying systemic disease. Vasculitis is considered to be the pathological mechanism<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a>; however, even though swollen endothelial cells and focal fibrinoid deposits were seen in the biopsy from our patient, it was not possible to make a conclusive diagnosis of cutaneous vasculitis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In a review of the literature, we have found that CS has rarely been associated with skin manifestations, and in most cases such manifestations are reported as rash or ulcers, with no further detailed clinical description or histopathology study. In a multicenter study that included 32 patients with CS, only 7 showed skin and mucous membrane involvement or chondritis: 3 cases of rash, with evidence of vasculitis in only 2 of them, 1 patient with photosensitivity, 1 with vitiligo, 2 with oral ulcers and 2 with chondritis.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Pagnini et al.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> described 23 children with CS; only 3 presented skin manifestations, all of which were described as rashes. As far as we are aware, these multiple ulcerated necrotic lesions have not previously been described in association with CS.</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary, we have described an unusual case of multiple ulcerated necrotic lesions in a patient with CS.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interests.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 1129 "Ancho" => 1600 "Tamanyo" => 165834 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Ulcerated necrotic lesions on both lower legs. (B) Linear ulcer on the left arm exhibiting the pathergy phenomenon.</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" => 616 "Ancho" => 1600 "Tamanyo" => 270328 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Ulcerated epidermis with a diffuse inflammatory infiltrate in the dermis. (B) Infiltrates formed of neutrophils, histiocytes, lymphocytes, and occasional multinucleated giant cells.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clinical features and outcome of Cogan syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "I. Pagnini" 1 => "M.E. Zannin" 2 => "F. Vittadello" 3 => "M. Sari" 4 => "G. Simonini" 5 => "R. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 8 | 10 | 18 |
| 2024 October | 82 | 44 | 126 |
| 2024 September | 73 | 28 | 101 |
| 2024 August | 96 | 46 | 142 |
| 2024 July | 81 | 32 | 113 |
| 2024 June | 68 | 38 | 106 |
| 2024 May | 53 | 34 | 87 |
| 2024 April | 79 | 29 | 108 |
| 2024 March | 65 | 26 | 91 |
| 2024 February | 65 | 34 | 99 |
| 2024 January | 52 | 36 | 88 |
| 2023 December | 54 | 19 | 73 |
| 2023 November | 71 | 29 | 100 |
| 2023 October | 56 | 29 | 85 |
| 2023 September | 48 | 31 | 79 |
| 2023 August | 48 | 17 | 65 |
| 2023 July | 52 | 31 | 83 |
| 2023 June | 40 | 27 | 67 |
| 2023 May | 36 | 37 | 73 |
| 2023 April | 26 | 33 | 59 |
| 2023 March | 51 | 23 | 74 |
| 2023 February | 56 | 18 | 74 |
| 2023 January | 51 | 34 | 85 |
| 2022 December | 52 | 47 | 99 |
| 2022 November | 35 | 27 | 62 |
| 2022 October | 48 | 27 | 75 |
| 2022 September | 66 | 34 | 100 |
| 2022 August | 48 | 32 | 80 |
| 2022 July | 45 | 29 | 74 |
| 2022 June | 37 | 21 | 58 |
| 2022 May | 28 | 28 | 56 |
| 2022 April | 31 | 35 | 66 |
| 2022 March | 37 | 36 | 73 |
| 2022 February | 39 | 35 | 74 |
| 2022 January | 52 | 34 | 86 |
| 2021 December | 42 | 42 | 84 |
| 2021 November | 41 | 38 | 79 |
| 2021 October | 44 | 54 | 98 |
| 2021 September | 46 | 32 | 78 |
| 2021 August | 32 | 36 | 68 |
| 2021 July | 23 | 25 | 48 |
| 2021 June | 25 | 28 | 53 |
| 2021 May | 28 | 50 | 78 |
| 2021 April | 44 | 26 | 70 |
| 2021 March | 59 | 16 | 75 |
| 2021 February | 46 | 23 | 69 |
| 2021 January | 23 | 13 | 36 |
| 2020 December | 22 | 19 | 41 |
| 2020 November | 13 | 16 | 29 |
| 2020 October | 23 | 28 | 51 |
| 2020 September | 20 | 20 | 40 |
| 2020 August | 18 | 14 | 32 |
| 2020 July | 12 | 13 | 25 |
| 2020 June | 21 | 23 | 44 |
| 2020 May | 13 | 12 | 25 |
| 2020 April | 14 | 8 | 22 |
| 2020 March | 17 | 17 | 34 |
| 2020 February | 8 | 0 | 8 |
| 2019 September | 1 | 0 | 1 |
| 2019 August | 3 | 0 | 3 |
| 2019 May | 0 | 1 | 1 |
| 2019 April | 0 | 3 | 3 |
| 2018 December | 5 | 0 | 5 |
| 2018 November | 4 | 0 | 4 |
| 2018 October | 9 | 0 | 9 |
| 2018 September | 9 | 0 | 9 |
| 2018 February | 42 | 2 | 44 |
| 2018 January | 38 | 7 | 45 |
| 2017 December | 87 | 7 | 94 |
| 2017 November | 33 | 1 | 34 |
| 2017 October | 34 | 5 | 39 |
| 2017 September | 37 | 8 | 45 |
| 2017 August | 29 | 9 | 38 |
| 2017 July | 27 | 6 | 33 |
| 2017 June | 46 | 6 | 52 |
| 2017 May | 59 | 27 | 86 |
| 2017 April | 8 | 3 | 11 |
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