Case Report
Chronic anterior uveitis in a patient with CREST syndrome
Uveítis anterior crónica en paciente con síndrome de CREST
Sara Fuente Cosíoa,
, Beatriz Arca Barcab, Paula Martínez Garcíac, Antonio Sampedro Lópezd
Corresponding author
a Medicina Interna, Hospital Comarcal de Jarrio, Coaña, Asturias, Spain
b Reumatología, Hospital Universitario San Agustín, Avilés, Asturias, Spain
c Medicina Interna, Hospital Universitario San Agustín, Avilés, Asturias, Spain
d Oftalmología, Hospital Universitario San Agustín, Avilés, Asturias, Spain
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LcSSc is frequently associated with CREST syndrome (Calcinosis cutis, Raynaud's phenomenon, Esophageal dysmotility, Sclerodactyly and Telangiectasia).<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> In this syndrome, ocular involvement is infrequent, the case presented below is the fourth described in the literature on uveitis with CREST syndrome.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3–5</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Description of the case</span><p id="par0010" class="elsevierStylePara elsevierViewall">A 53-year-old female, housewife, no family history of interest, no known drug allergies or toxic habits, with a history of Hashimoto's thryoiditis and multinodular goiter. Followed up by rheumatology for CREST syndrome and secondary Sjögren's syndrome, under treatment with nifedipine, esomeprazole, cinitapride and levothyroxine.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Complementary studies showed titre positive ANA (1/2560) centromere pattern and capillaroscopy with sclerodermal pattern. No anomalies were observed on chest X-rays, functional, respiratory and diffusion tests or transthoracic echocardiograph.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The patient attended ophthalmology consultations for repeated episodes of red eye, pain and loss of right eye vision. On examination, she presented a non-hyperaemic eye with some isolated cells inside the anterior chamber (Tyndall 1<span class="elsevierStyleSup">+</span> and negative flare),<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> synechias and pigment deposits in anterior crystalloids, together with cystoid macular oedema with visual acuity (VA) .5 in the right eye (RE); and was diagnosed with chronic anterior uveitis with secondary macular oedema. She was prescribed cycloplegic and topical corticosteroids (dexamethasone eye drops and subtenon's injection of triamcinolone acetonide) with progressive improvement. The patient continued to experience new flare-ups, coinciding with decrease and/or withdrawal of the corticosteroid; and therefore in approximately one year, oral methotrexate was started (15<span class="elsevierStyleHsp" style=""></span>mg/week). After 5 months it was necessary to increase the dose (20<span class="elsevierStyleHsp" style=""></span>mg/week/sc) and then discontinue it due to elevated transaminases after 2 months of treatment. She has remained stable since that time, 5 further flare-ups presented after that, and she started oral cyclosporine (50<span class="elsevierStyleHsp" style=""></span>mg/12<span class="elsevierStyleHsp" style=""></span>h) with improvement and clinical stabilisation until now (VA .7 in RE and .8 in LE).</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion of the case</span><p id="par0025" class="elsevierStylePara elsevierViewall">Uveitis consists of inflammation of the uvea, the middle portion of the eye, comprising the iris and the ciliary body (anterior part) and choroid (posterior part); although, in general terms, it includes any eye inflammation. Its principal aetiologies are: infections, immune-mediated systemic diseases (40%), syndromes confined to the eye and idiopathic (up to 30%). Most notable among the immune-mediated aetiologies are spondyloarthritis, sarcoidosis, Behçet's syndrome and inflammatory bowel disease, among others.</p><p id="par0030" class="elsevierStylePara elsevierViewall">As for the ocular manifestations of SSc, the most notable are the palpebral alterations due to fibrotic changes, which occur more frequently in young patients, with greater skin involvement and earlier onset of the disease. Other ocular manifestations frequently related to this disease are: pinguecula, keratoconjunctivitis sicca, cataracts and blepharitis. Inflammatory ocular manifestations have been described, although much less frequently: of interest is the recent description of 2 cases of CREST syndrome with retinal vasculitis.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> We should also mention that in the study by Gomes et al.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> only 4.4% (2 patients) episcleritis was found and no cases of uveitis were described. In fact, this combination (uveitis and SSc) is very rare and only isolated cases have been described in the literature: 3 in patients with CREST syndrome,<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3–5</span></a> one in a patient with SSc<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> and another in a patient with SSc without sclerodermia.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> although the frequency of this association is low, we believe that it should be taken into consideration when managing these situations in order to deliver multidisciplinary treatment in which ophthalmologists would play an important role.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1420645" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1299100" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1420646" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1299101" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Description of the case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion of the case" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-07-11" "fechaAceptado" => "2018-10-18" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1299100" "palabras" => array:3 [ 0 => "CREST syndrome" 1 => "Systemic sclerosis" 2 => "Uveitis" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1299101" "palabras" => array:3 [ 0 => "Síndrome de CREST" 1 => "Esclerosis sistémica" 2 => "Uveítis" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Systemic sclerosis is a connective tissue pathology with very heterogeneous clinical manifestations, associated in a small percentage with inflammatory eye diseases. In the specific case of uveitis, only isolated cases have been reported in the literature, especially in relation to the CREST syndrome. We present the case of a 53-year-old woman with CREST syndrome and chronic anterior uveitis, which we consider of clinical relevance given its low prevalence.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La esclerosis sistémica es una conectivopatía con unas manifestaciones clínicas muy heterogéneas, relacionándose en un pequeño porcentaje con enfermedades oculares inflamatorias. En el caso concreto de las uveítis, únicamente se han descrito casos aislados en la literatura, sobre todo con relación al síndrome de CREST. Presentamos el caso de una mujer de 53 años, con síndrome de CREST y uveítis anterior crónica, que consideramos de relevancia clínica dada su baja prevalencia.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Fuente Cosío S, Arca Barca B, Martínez García P, Sampedro López A. Uveítis anterior crónica en paciente con síndrome de CREST. Reumatol Clin. 2020;16:497–498.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pathogenesis of systemic sclerosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "J.H. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 6 | 9 |
| 2024 October | 50 | 37 | 87 |
| 2024 September | 52 | 32 | 84 |
| 2024 August | 69 | 54 | 123 |
| 2024 July | 61 | 38 | 99 |
| 2024 June | 55 | 31 | 86 |
| 2024 May | 45 | 27 | 72 |
| 2024 April | 53 | 29 | 82 |
| 2024 March | 52 | 32 | 84 |
| 2024 February | 51 | 26 | 77 |
| 2024 January | 49 | 12 | 61 |
| 2023 December | 59 | 26 | 85 |
| 2023 November | 61 | 40 | 101 |
| 2023 October | 38 | 32 | 70 |
| 2023 September | 79 | 48 | 127 |
| 2023 August | 74 | 11 | 85 |
| 2023 July | 36 | 25 | 61 |
| 2023 June | 30 | 23 | 53 |
| 2023 May | 30 | 29 | 59 |
| 2023 April | 38 | 20 | 58 |
| 2023 March | 46 | 38 | 84 |
| 2023 February | 30 | 32 | 62 |
| 2023 January | 34 | 20 | 54 |
| 2022 December | 60 | 50 | 110 |
| 2022 November | 40 | 39 | 79 |
| 2022 October | 66 | 49 | 115 |
| 2022 September | 42 | 55 | 97 |
| 2022 August | 37 | 47 | 84 |
| 2022 July | 36 | 48 | 84 |
| 2022 June | 34 | 43 | 77 |
| 2022 May | 41 | 58 | 99 |
| 2022 April | 46 | 46 | 92 |
| 2022 March | 48 | 75 | 123 |
| 2022 February | 53 | 46 | 99 |
| 2022 January | 44 | 47 | 91 |
| 2021 December | 37 | 50 | 87 |
| 2021 November | 49 | 48 | 97 |
| 2021 October | 96 | 79 | 175 |
| 2021 September | 54 | 47 | 101 |
| 2021 August | 45 | 37 | 82 |
| 2021 July | 35 | 28 | 63 |
| 2021 June | 34 | 34 | 68 |
| 2021 May | 35 | 42 | 77 |
| 2020 May | 1 | 2 | 3 |
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