Case Report
Association between three autoimmune diseases: vitiligo, primary biliary cirrhosis, and Sjögren's syndrome
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true ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Pinar Incel Uysal, Neslihan Akdogan, Yildiz Hayran, Ayse Oktem, Basak Yalcin" "autores" => array:5 [ 0 => array:2 [ "nombre" => "Pinar" "apellidos" => "Incel Uysal" ] 1 => array:2 [ "nombre" => "Neslihan" "apellidos" => "Akdogan" ] 2 => array:2 [ "nombre" => "Yildiz" "apellidos" => "Hayran" ] 3 => array:2 [ "nombre" => "Ayse" "apellidos" => "Oktem" ] 4 => array:2 [ "nombre" => "Basak" "apellidos" => "Yalcin" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "pt" => array:9 [ "pii" => "S2666275219300591" "doi" => "10.1016/j.abdp.2019.03.002" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "pt" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2666275219300591?idApp=UINPBA00008Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0365059619300704?idApp=UINPBA00008Z" "url" => "/03650596/0000009400000006/v1_202001030404/S0365059619300704/v1_202001030404/en/main.assets" ] "en" => array:19 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>" "titulo" => "Association between three autoimmune diseases: vitiligo, primary biliary cirrhosis, and Sjögren's syndrome" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "710" "paginaFinal" => "712" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Lara Silveira Abdo-Aguiar, Caio César Silva de Castro" "autores" => array:2 [ 0 => array:4 [ "nombre" => "Lara Silveira" "apellidos" => "Abdo-Aguiar" "email" => array:1 [ 0 => "lara_abdo@yahoo.com.br" ] "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" => "Caio César" "apellidos" => "Silva de Castro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0025" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Dermatology Service, Hospital Santa Casa de Misericórdia de Curitiba, Curitiba, PR, Brazil" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Discipline of Dermatology, Pontifícia Universidade Católica do Paraná, Curitiba, PR, Brazil" "etiqueta" => "b" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "?" "correspondencia" => "Corresponding author." ] ] ] ] "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" => 981 "Ancho" => 905 "Tamanyo" => 106494 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Vitiligo on left hemiface (Wood's light examination).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Vitiligo is a chronic autoimmune disease characterized by the appearance of hypochromic and achromic macules and patches on the skin and mucous membranes, due to the disappearance of melanocytes in the affected area.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In turn, primary biliary cirrhosis (PBC) is a chronic hepatic autoimmune disease characterized by the destruction of the epithelial cell lining the intrahepatic bile ducts, progressively evolving to fibrosis and cirrhosis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Sjogren's syndrome (SS) is a chronic autoimmune condition affecting exocrine glands, especially the lacrimal and salivary glands. The main clinical manifestations are xerostomia and xerophthalmia. The primary form of the disease is more prevalent in females than males, in a ratio of 9:1, affecting approximately 0.5% of the population, a prevalence similar to that of systemic lupus erythematosus and systemic sclerosis.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It is well known that the occurrence of an autoimmune disease increases the risk of other autoimmune diseases in the same patient, a phenomenon that has been explored in previously published reports and case series; however, the association between common vitiligo, PBC and SS has not been published to date.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A female patient was diagnosed with PBC and SS at 54 years of age. She started treatment with ursodeoxycholic acid, deflazacort, hydroxychloroquine, and pilocarpine, reaching satisfactory disease control. Subsequently, at age 58, she evolved with hypochromic and, later, achromic lesions characteristic of vitiligo restricted to the facial area (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>), associated with significant impairment in self-esteem and quality of life. She had a positive family history of vitiligo in a third-degree relative. At the first evaluation, the patient had negative antinuclear antibody (ANA) and anemia due to chronic iron deficiency, requiring blood transfusion. For vitiligo, topical treatment with tacrolimus and phototherapy with narrow band UVB were indicated, which stabilized the progression of the disease, without new lesions appearing on the face or other areas of the skin. After starting phototherapy, laboratory tests showed positive ANA with mixed pattern – thick reticulated speckled 1:320 and fine speckled 1:640 – with no other changes.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Autoimmune diseases are chronic conditions that result from the loss of immune tolerance to self-antigens. The origin of autoimmunity mechanisms remains partially known, and a combination of genetic, immunological, environmental, and hormonal factors is linked to its development. Autoimmune diseases can be classified as either organ-specific (<span class="elsevierStyleItalic">e.g.</span>, myasthenia gravis, Graves’ disease, and polymyositis) or multisystem diseases (<span class="elsevierStyleItalic">e.g.</span>, systemic lupus erythematosus, rheumatoid arthritis, and systemic sclerosis).<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Vitiligo is a cutaneous disease of chronic and autoimmune nature that, although occurring sporadically, is known to have a certain degree of heredity. Studies have previously demonstrated the involvement of different genes in its pathogenesis, but to date there is no known evidence of a validated serum or tissue marker associated with its occurrence. Case reports and series have associated vitiligo with other autoimmune diseases, and studies have reported common genes between this condition and systemic lupus erythematosus and Hashimoto's thyroiditis.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">PBC is considered a prototype of autoimmune disease due to the characteristic antimitochondrial autoantibody, its homogeneous clinical presentation, and the specificity of the anatomopathological findings. Genetic deficiencies and mechanisms of immune regulation take part in the pathogenesis of the disease, which results in damage exclusively to the small- and medium-caliber bile duct cells; over time, consequently and progressively, a state of chronic autoimmune cholangitis develops.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In 2012, Efe et al. carried out a multicenter study of 71 patients with PBC and autoimmune hepatitis (AIH) to analyze the association of these with other autoimmune diseases. The data indicated that 76.1% of the patients had a positive anti-nuclear factor, 74.6% had antimitochondrial antibodies, and 52.1% had both markers. In 31 patients (43.6%), 14 extrahepatic autoimmune comorbidities were identified, with predominance for thyroid disorders in 13 participants of the study. Other findings included SS in six participants (8.4%) and psoriasis, celiac disease, and rheumatoid arthritis in three patients each (4.2%). Vitiligo was observed in two patients (2.8%); the same proportion was observed for systemic lupus erythematosus.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The concomitant occurrence of multiple autoimmune diseases in the same patient draws attention to the presence of common mechanisms. The concept of mosaic of autoimmunity has been proposed to describe this condition and demonstrated in reports and series of cases, as mentioned above, although the specific immunological and genetic mechanisms have not yet been fully elucidated.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Patients with autoimmune hepatic diseases, such as PBC and AIH, may also present with other organ-specific or multisystemic autoimmune conditions. In the aforementioned individuals, SS was the most frequent multisystem autoimmune comorbidity.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Two previously published studies analyzed 34 SS patients who had a concomitant elevation of hepatic enzymes, among which a diagnosis of PBC or AIH was made in 15 and nine patients, respectively.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">A significant portion of patients with PBC suffer from the so-called sicca syndrome, and some of them present classical SS. Both PBC and SS are characterized by inflammation and immune-mediated destruction of epithelial tissue; moreover, the PDC-E2 target antigen has been identified in both bile duct and salivary gland epithelium, which strongly demonstrates the association between both conditions.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,5</span></a> In addition, the finding that the salivary and lacrimal glands, as well as the urinary tract epithelium, may also be damaged in PBC has led to the hypothesis that PBC, similarly to SS, can be considered a generalized epithelitis.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> To date, there is no evidence that vitiligo has the same pathophysiological mechanism.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Finally, the association between this patient's comorbidities and their underlying pathophysiological mechanism was clarified; the triple association was explained by the mosaic of autoimmunity and the occurrence of epithelitis.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0055" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Author's contributions</span><p id="par0060" class="elsevierStylePara elsevierViewall">Lara Silveira Abdo Aguiar: Elaboration and writing of the manuscript; obtaining, analyzing and interpreting the data; intellectual participation in propaedeutic and/or therapeutic conduct of the cases studied.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Caio César Silva de Castro: Conception and planning of the study; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of the cases studied; critical review of the literature.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1282773" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1185779" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 3 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 4 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 5 => array:2 [ "identificador" => "sec0020" "titulo" => "Financial support" ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Author's contributions" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-03-20" "fechaAceptado" => "2018-07-24" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1185779" "palabras" => array:3 [ 0 => "Autoimmunity" 1 => "Liver cirrhosis, biliary" 2 => "Sjogren's syndrome, Vitiligo" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Although the association of multiple autoimmune diseases has already been widely described, no reports of the association between vitiligo, primary biliary cirrhosis and Sjogren's syndrome were retrieved in the SciELO and PubMed databases. The authors describe the case of a female patient who was diagnosed with primary biliary cirrhosis and Sjogren's syndrome at age 54. At age 58, she developed vitiligo restricted to the face, associated with significant impairment of self-esteem and quality of life. Antinuclear antibody was negative at the onset of the condition, but became positive after phototherapy initiation. In general, the occurrence of multiple autoimmune diseases in the same patient is known as a mosaic of autoimmunity. However, specific mechanisms appear to interconnect primary biliary cirrhosis and Sjogren's syndrome, such as PDC-E2-mediated generalized epithelitis.</p></span>" ] ] "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "?" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Abdo-Aguiar LS, Castro CCS. Association between three autoimmune diseases: vitiligo, primary biliary cirrhosis and Sjögren's syndrome. An Bras Dermatol. 2019;94:710–2.</p>" ] 1 => array:2 [ "etiqueta" => "??" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital Santa Casa de Misericórdia de Curitiba, Curitiba, PR, Brazil.</p>" ] ] "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" => 874 "Ancho" => 905 "Tamanyo" => 92688 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Vitiligo on the face, front position (Wood's light examination).</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" => 981 "Ancho" => 905 "Tamanyo" => 106494 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Vitiligo on left hemiface (Wood's light examination).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Vitiligo – part 1" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "R.G. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 57 | 27 | 84 |
| 2024 September | 151 | 97 | 248 |
| 2024 August | 149 | 116 | 265 |
| 2024 July | 163 | 152 | 315 |
| 2024 June | 129 | 82 | 211 |
| 2024 May | 95 | 64 | 159 |
| 2024 April | 127 | 119 | 246 |
| 2024 March | 97 | 73 | 170 |
| 2024 February | 125 | 78 | 203 |
| 2024 January | 100 | 61 | 161 |
| 2023 December | 105 | 55 | 160 |
| 2023 November | 95 | 83 | 178 |
| 2023 October | 63 | 73 | 136 |
| 2023 September | 83 | 70 | 153 |
| 2023 August | 86 | 42 | 128 |
| 2023 July | 81 | 30 | 111 |
| 2023 June | 74 | 39 | 113 |
| 2023 May | 63 | 28 | 91 |
| 2023 April | 47 | 24 | 71 |
| 2023 March | 98 | 57 | 155 |
| 2023 February | 54 | 32 | 86 |
| 2023 January | 44 | 52 | 96 |
| 2022 December | 51 | 31 | 82 |
| 2022 November | 56 | 68 | 124 |
| 2022 October | 75 | 54 | 129 |
| 2022 September | 48 | 56 | 104 |
| 2022 August | 46 | 58 | 104 |
| 2022 July | 45 | 56 | 101 |
| 2022 June | 55 | 60 | 115 |
| 2022 May | 47 | 57 | 104 |
| 2022 April | 60 | 51 | 111 |
| 2022 March | 56 | 68 | 124 |
| 2022 February | 40 | 29 | 69 |
| 2022 January | 46 | 70 | 116 |
| 2021 December | 43 | 57 | 100 |
| 2021 November | 42 | 60 | 102 |
| 2021 October | 46 | 70 | 116 |
| 2021 September | 41 | 60 | 101 |
| 2021 August | 47 | 55 | 102 |
| 2021 July | 34 | 54 | 88 |
| 2021 June | 34 | 49 | 83 |
| 2021 May | 47 | 79 | 126 |
| 2021 April | 119 | 193 | 312 |
| 2021 March | 75 | 67 | 142 |
| 2021 February | 44 | 25 | 69 |
| 2021 January | 38 | 15 | 53 |
| 2020 December | 26 | 16 | 42 |
| 2020 November | 35 | 18 | 53 |
| 2020 October | 32 | 12 | 44 |
| 2020 September | 22 | 11 | 33 |
| 2020 August | 21 | 10 | 31 |
| 2020 July | 10 | 8 | 18 |
| 2020 June | 10 | 15 | 25 |
| 2020 May | 27 | 10 | 37 |
| 2020 April | 2 | 2 | 4 |
| 2020 March | 5 | 1 | 6 |
| 2020 February | 9 | 8 | 17 |
| 2020 January | 6 | 2 | 8 |
| 2019 December | 0 | 6 | 6 |
| 2019 November | 0 | 6 | 6 |
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