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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cogan syndrome &#40;CS&#41; is a rare vasculitis characterized by recurrent interstitial keratitis and audiovestibular symptoms including hearing loss&#44; tinnitus&#44; and vertigo&#46; To date&#44; cutaneous lesions have been only rarely described associated with this autoimmune disease&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> We present the case of a patient with CS and multiple ulcerated lesions&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 39-year-old white Spanish woman diagnosed with CS at age 31 years&#44; with stromal keratitis and severe bilateral hearing loss&#44; consulted for a 2-week history of ulcers that had started as pustules on the legs and then spread to the thighs&#44; arms&#44; abdomen&#44; and back&#46; At the time of onset&#44; the patient was on treatment with methotrexate &#40;25<span class="elsevierStyleHsp" style=""></span>mg&#47;wk&#41;&#44; prednisone &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#44; and tozilizumab &#40;8<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;mo&#41;&#46; Physical examination revealed numerous pustules and ulcerated necrotic lesions on the legs&#44; thighs&#44; arms&#44; back&#44; and abdomen &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; On suspicion of ecthyma&#44; topical antibiotics and oral cloxacillin were prescribed&#46; Smear cultures for bacteria&#44; fungi&#44; and mycobacteria were all negative&#46; Additional tests were within normal limits and the chest radiograph was normal&#46; Two weeks later the patient presented further pustules and enlargement of those previously present&#46; At no time did she report fever or malaise&#46; Histopathology of a biopsy from an ulcer on the left thigh revealed an ulcerated epidermis with epidermal necrosis and pustules at the border&#44; with an abscessified area of skin and a diffuse dermal inflammatory infiltrate formed by neutrophils&#44; histiocytes&#44; lymphocytes&#44; and occasional multinucleated giant cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41;&#46; No clear signs of vasculitis were observed&#46; Immunofluorescence was negative&#46; Bacterial cultures from the cutaneous biopsy and new smears were positive for <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span>&#47;<span class="elsevierStyleItalic">Staphylococcus haemolyticus</span>&#44; and <span class="elsevierStyleItalic">Streptococcus pyogenes</span>&#47;<span class="elsevierStyleItalic">Pseudomonas putida fluorescens</span>&#44; respectively&#46; Oral cefuroxime and ciprofloxacin were started but the lesions showed no improvement&#46; It was then that prednisone &#40;at a dose of up to 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d&#41; was prescribed&#46; The patient responded favorably&#44; 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&#44; suggesting a pathergy phenomenon &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; At follow-up&#44; no lesions were observed&#46; At the time of writing&#44; the patient remains asymptomatic and continues her usual therapy&#46;</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&#44; and ecthyma was our initial provisional diagnosis&#46; The lack of response&#8212;and even a deterioration&#8212;of the lesions after antibiotic therapy&#44; the pathergy phenomenon&#44; and the favorable response to steroids led us consider a pyoderma gangrenosum &#40;PG&#41;-like neutrophilic dermatosis&#46; However&#44; the multiple lesions and the histopathology were not conclusive for either PG or other forms of neutrophilic dermatosis&#46; Multiple lesions have rarely been described in PG&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> In 70&#37; of cases&#44; PG is associated with an underlying disease such as inflammatory bowel disease &#40;IBD&#41;&#44; inflammatory arthritis&#44; or a hematologic malignancy&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Tirelli et al&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> found about 250 reports of patients with CS&#44; only 13 of whom had concomitant chronic IBD&#59; of these&#44; none experienced improvement after therapy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of CS are unknown&#46; Initially&#44; the disease was thought to be caused by an infection&#44; but it is now considered to be an autoimmune disorder&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In addition to the ocular and audiovestibular involvement&#44; numerous systemic manifestations have been reported in CS&#44; most commonly of cardiovascular&#44; neurological&#44; or gastrointestinal origin&#46; Approximately 70&#37; of patients have an underlying systemic disease&#46; Vasculitis is considered to be the pathological mechanism<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a>&#59; however&#44; even though swollen endothelial cells and focal fibrinoid deposits were seen in the biopsy from our patient&#44; it was not possible to make a conclusive diagnosis of cutaneous vasculitis&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">In a review of the literature&#44; we have found that CS has rarely been associated with skin manifestations&#44; and in most cases such manifestations are reported as rash or ulcers&#44; with no further detailed clinical description or histopathology study&#46; In a multicenter study that included 32 patients with CS&#44; only 7 showed skin and mucous membrane involvement or chondritis&#58; 3 cases of rash&#44; with evidence of vasculitis in only 2 of them&#44; 1 patient with photosensitivity&#44; 1 with vitiligo&#44; 2 with oral ulcers and 2 with chondritis&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Pagnini et al&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> described 23 children with CS&#59; only 3 presented skin manifestations&#44; all of which were described as rashes&#46; As far as we are aware&#44; these multiple ulcerated necrotic lesions have not previously been described in association with CS&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary&#44; we have described an unusual case of multiple ulcerated necrotic lesions in a patient with CS&#46;</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&#46;</p></span></span>"
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Case and Research Letter
Atypical Ulcerated Lesions in a Patient With Cogan Syndrome
Lesiones ulcerativas atípicas en un paciente con síndrome de Cogan
G. Solano-Lópeza,
Autor para correspondencia
guitje1@hotmail.com

Corresponding author.
, A. Montes-Torresa, E. Tomerob, J. Fragac, D. De Argilaa, E. Daudéna
a Department of Dermatology, Hospital Universitario de la Princesa, Madrid, Spain
b Department of Rheumatology, Hospital Universitario de la Princesa, Madrid, Spain
c Department of Pathology, Hospital Universitario de la Princesa, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cogan syndrome &#40;CS&#41; is a rare vasculitis characterized by recurrent interstitial keratitis and audiovestibular symptoms including hearing loss&#44; tinnitus&#44; and vertigo&#46; To date&#44; cutaneous lesions have been only rarely described associated with this autoimmune disease&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> We present the case of a patient with CS and multiple ulcerated lesions&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 39-year-old white Spanish woman diagnosed with CS at age 31 years&#44; with stromal keratitis and severe bilateral hearing loss&#44; consulted for a 2-week history of ulcers that had started as pustules on the legs and then spread to the thighs&#44; arms&#44; abdomen&#44; and back&#46; At the time of onset&#44; the patient was on treatment with methotrexate &#40;25<span class="elsevierStyleHsp" style=""></span>mg&#47;wk&#41;&#44; prednisone &#40;10<span class="elsevierStyleHsp" style=""></span>mg&#47;d&#41;&#44; and tozilizumab &#40;8<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;mo&#41;&#46; Physical examination revealed numerous pustules and ulcerated necrotic lesions on the legs&#44; thighs&#44; arms&#44; back&#44; and abdomen &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; On suspicion of ecthyma&#44; topical antibiotics and oral cloxacillin were prescribed&#46; Smear cultures for bacteria&#44; fungi&#44; and mycobacteria were all negative&#46; Additional tests were within normal limits and the chest radiograph was normal&#46; Two weeks later the patient presented further pustules and enlargement of those previously present&#46; At no time did she report fever or malaise&#46; Histopathology of a biopsy from an ulcer on the left thigh revealed an ulcerated epidermis with epidermal necrosis and pustules at the border&#44; with an abscessified area of skin and a diffuse dermal inflammatory infiltrate formed by neutrophils&#44; histiocytes&#44; lymphocytes&#44; and occasional multinucleated giant cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41;&#46; No clear signs of vasculitis were observed&#46; Immunofluorescence was negative&#46; Bacterial cultures from the cutaneous biopsy and new smears were positive for <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span>&#47;<span class="elsevierStyleItalic">Staphylococcus haemolyticus</span>&#44; and <span class="elsevierStyleItalic">Streptococcus pyogenes</span>&#47;<span class="elsevierStyleItalic">Pseudomonas putida fluorescens</span>&#44; respectively&#46; Oral cefuroxime and ciprofloxacin were started but the lesions showed no improvement&#46; It was then that prednisone &#40;at a dose of up to 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;d&#41; was prescribed&#46; The patient responded favorably&#44; 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&#44; suggesting a pathergy phenomenon &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; At follow-up&#44; no lesions were observed&#46; At the time of writing&#44; the patient remains asymptomatic and continues her usual therapy&#46;</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&#44; and ecthyma was our initial provisional diagnosis&#46; The lack of response&#8212;and even a deterioration&#8212;of the lesions after antibiotic therapy&#44; the pathergy phenomenon&#44; and the favorable response to steroids led us consider a pyoderma gangrenosum &#40;PG&#41;-like neutrophilic dermatosis&#46; However&#44; the multiple lesions and the histopathology were not conclusive for either PG or other forms of neutrophilic dermatosis&#46; Multiple lesions have rarely been described in PG&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> In 70&#37; of cases&#44; PG is associated with an underlying disease such as inflammatory bowel disease &#40;IBD&#41;&#44; inflammatory arthritis&#44; or a hematologic malignancy&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Tirelli et al&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> found about 250 reports of patients with CS&#44; only 13 of whom had concomitant chronic IBD&#59; of these&#44; none experienced improvement after therapy&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of CS are unknown&#46; Initially&#44; the disease was thought to be caused by an infection&#44; but it is now considered to be an autoimmune disorder&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> In addition to the ocular and audiovestibular involvement&#44; numerous systemic manifestations have been reported in CS&#44; most commonly of cardiovascular&#44; neurological&#44; or gastrointestinal origin&#46; Approximately 70&#37; of patients have an underlying systemic disease&#46; Vasculitis is considered to be the pathological mechanism<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a>&#59; however&#44; even though swollen endothelial cells and focal fibrinoid deposits were seen in the biopsy from our patient&#44; it was not possible to make a conclusive diagnosis of cutaneous vasculitis&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">In a review of the literature&#44; we have found that CS has rarely been associated with skin manifestations&#44; and in most cases such manifestations are reported as rash or ulcers&#44; with no further detailed clinical description or histopathology study&#46; In a multicenter study that included 32 patients with CS&#44; only 7 showed skin and mucous membrane involvement or chondritis&#58; 3 cases of rash&#44; with evidence of vasculitis in only 2 of them&#44; 1 patient with photosensitivity&#44; 1 with vitiligo&#44; 2 with oral ulcers and 2 with chondritis&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Pagnini et al&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> described 23 children with CS&#59; only 3 presented skin manifestations&#44; all of which were described as rashes&#46; As far as we are aware&#44; these multiple ulcerated necrotic lesions have not previously been described in association with CS&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">In summary&#44; we have described an unusual case of multiple ulcerated necrotic lesions in a patient with CS&#46;</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&#46;</p></span></span>"
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ISSN: 00017310
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