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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Sarcoidosis is a chronic&#44; idiopathic multisystemic granulomatous disease&#46; The incidence varies widely throughout the world&#44; probably because of differences in environmental exposures&#44; predisposing HLA alleles and other genetic factors&#46; The higher incidence has been observed in northern European countries and the adjusted annual incidence among black Americans is approximately three times that among Caucasian Americans&#44; with a preponderance of cases in females across racial and ethnic groups&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Sarcoidal granulomas can involve any organ&#44; but in more than 90&#37; of patients is manifested as intrathoratic lymph-node enlargement and pulmonary involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> Extrapulmonary organ involvement occurs in up to 50&#37; of patients&#44; while only 5&#8211;9&#37; have extrapulmonary disease without pulmonary sarcoidosis&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> In regards to specific organ involvement&#44; the non-pulmonary sarcoidosis patients have a higher prevalence of skin involvement &#40;49&#46;3&#37;&#41; in comparison to those with pulmonary sarcoidosis &#40;25&#37;&#41;&#46; After skin&#44; the distribution by organ involvement in those patients with non-pulmonary sarcoidosis is eye &#40;23&#46;6&#37;&#41;&#44; liver &#40;20&#46;7&#37;&#41;&#44; extrathoracic lymph-node &#40;13&#46;7&#37;&#41;&#44; parotid&#47;salivary &#40;5&#46;7&#37;&#41; and bone&#47;joint &#40;1&#46;4&#37;&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Criteria for diagnosing systemic sarcoidosis require symptoms showing the involvement of at least two organ systems&#44; supporting histology and valid exclusion of other granulomatous disease&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Multi-organ involvement in non-pulmonary sarcoidosis affecting more than three organs are rarely reported and constitute a clinical challenge&#46; There are scarce literature data published on diagnosis&#44; clinical course and management in those cases&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case presentation</span><p id="par0025" class="elsevierStylePara elsevierViewall">A 41-year-old Turkish woman&#44; was referred to our department presenting severe asthenia&#44; xerophthalmia&#44; xerostomia&#44; inflammatory arthralgias and recurrent bilateral parotid enlargement&#46; On physical examination&#44; the eversion of the upper eyelids showed a lacrimal gland enlargement &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#44; arrow&#41;&#46; Supraclavicular lymph nodes were enlarged as well and palpable&#46; There were no skin lesions at any anatomical area&#46; The initial blood test showed an increase in C-reactive protein of 1&#46;03<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#40;&#60;0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41; associated to elevated liver enzymes with a total bilirubin of 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;dL &#40;0&#46;2&#8211;1&#46;2<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#44; AST&#47;GOT 56<span class="elsevierStyleHsp" style=""></span>IU&#47;L &#40;0&#8211;32<span class="elsevierStyleHsp" style=""></span>IU&#47;L&#41;&#44; ALT&#47;GPT 75<span class="elsevierStyleHsp" style=""></span>IU&#47;L &#40;0&#8211;33<span class="elsevierStyleHsp" style=""></span>IU&#47;L&#41;&#44; GGT 112<span class="elsevierStyleHsp" style=""></span>IU&#47;L &#40;5&#8211;36<span class="elsevierStyleHsp" style=""></span>IU&#47;L&#41;&#44; alkaline phosphatase 157<span class="elsevierStyleHsp" style=""></span>IU&#47;L &#40;35&#8211;104<span class="elsevierStyleHsp" style=""></span>IU&#47;L&#41;&#46; No alteration of the cell count or hypergammaglobulinemia were observed&#46; Infectious serologies including the most prevalent viral&#44; bacterial and fungal entities were negatives&#46; Antinuclear&#44; anti-SSA&#47;Ro&#44; antineutrophil cytoplasmic&#44; anti-myeloperoxidase and anti-proteinase 3 antibodies were all negatives&#46; A computed tomography &#40;CT&#41; of the thorax and abdomen was performed to rule out malignancy&#46; The thoracic CT scan did not show pulmonary parenchymal abnormalities and neither mediastinal or hilar lymphadenopathy&#46; The abdominal CT scan showed enlarged periceliac and periportal lymph nodes without hepatomegaly&#46; A lacrimal gland and supraclavicular lymph node biopsy were performed showing confluent epithelioid granulomas without necrosis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#42;&#41;&#46; On suspicion of sarcoidosis with involvement of the lacrimal glands and peripheral lymph nodes&#44; systemic corticosteroids &#40;GC&#41; were initiated at a dose of 0&#46;5<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#44; gradually taper during the following eight months to a maintenance dose of 15<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#46; During the follow-up and once the patient finished the systemic GC therapy&#44; isolated hepatic progression was observed with progressive and worsening of the liver function tests characterized by dissociated cholestasis and cytolysis&#46; Thus&#44; an hepatic biopsy was performed which supported the diagnosis of hepatic sarcoidosis &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; A new course of systemic GC was initiated at 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day gradually taper during the following ten months with fluctuation of the liver enzymes showing a mild dissociated cholestasis without cytolysis and with no hepatic sign or symptoms of disease progression&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion and conclusion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Sarcoidosis is a granulomatous systemic autoimmune disease of unknown and probably multifactorial aetiology&#59; whose typical histopathological finding is the presence of non-necrotizing granulomas&#46; Its prevalence is estimated between 0&#46;02 and 0&#46;14&#37;&#44; being more prevalent in Nordic countries and African-Americans&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1&#44;2</span></a> In addition&#44; sarcoidosis occurrence varies greatly by age and gender with a peak age of onset closer to 30&#8211;55 years-old&#44; earlier in men than in women&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">2&#44;4</span></a> The difference in age between gender at diagnosis suggests that could be related to genetic factors or environmental exposures&#44; although what causes this variation is unknown&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Between 5 and 9&#37; of patients with sarcoidosis have isolated extrapulmonary disease without pulmonary involvement&#46;<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3&#44;5</span></a> According to the affected organ&#44; ocular involvement is found in 23&#46;6&#37;&#44; parotid&#47;salivary gland in 5&#46;7&#37;&#44; hepatic involvement in 20&#46;7&#37; and extrathoracic lymphadenopathy in 13&#46;7&#37; of the patients with non-pulmonary sarcoidosis&#46;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">We report an uncommon presentation of non-pulmonary sarcoidosis with multi-organ involvement &#40;lacrimal gland&#44; peripheral lymph nodes and liver&#41;&#46; To our knowledge&#44; this is the first reported case of non-pulmonary sarcoidosis involving hepatic&#44; lymph nodes and lacrimal gland at the same time&#46; In addition&#44; our patient presented bilateral parotid gland enlargement&#59; however&#44; the biopsy was avoided to prevent iatrogenesis&#46; After a careful diagnostic work-up&#44; infectious agents&#44; drug-induced hypersensitivity&#44; connective tissue diseases such as vasculitis and Sj&#246;gren syndrome&#44; primary immunodeficiencies and sarcoid-like reaction to malignancy were excluded&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the prognosis of the disease will be marked by the liver therapeutic response&#46; The therapeutic approach for hepatic involvement should be focus in avoiding disease progression and hepatic complications&#46; The first-line treatment is based on systemic GC&#59; however&#44; some patients have not a complete clinical response&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> Alternative agents&#44; such as methotrexate&#44; despite the potential risk of hepatotoxicity has showed improve in the liver function test abnormalities and also&#44; it is helpful as corticosteroid sparing&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">6&#44;7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">The relative infrequency of sarcoidosis&#44; accompanied by its wide clinical heterogeneity&#44; leads to a complex diagnostic process that poses a great challenge in routine clinical practice&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">All named authors declare that they have no conflict of interest to disclose&#46;</p></span></span>"
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          "palabras" => array:6 [
            0 => "Sarcoidosis"
            1 => "Extrapulmonar"
            2 => "Enfermedad granulomatosa"
            3 => "Gl&#225;ndula lacrimal"
            4 => "Adenopat&#237;a perif&#233;rica"
            5 => "H&#237;gado"
          ]
        ]
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      "en" => array:2 [
        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Isolated extrapulmonary involvement in sarcoidosis is uncommon and reported in 5&#8211;9&#37; of systemic sarcoidosis&#44; this constitutes a clinical challenge due to its extensive differential diagnosis&#46; Extrapulmonary sarcoidosis affecting more than three organs is rarely reported and there are scarce literature data published on diagnosis&#44; clinical course and management in those cases&#46;</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We hereby discuss a case of a 41-year-old female with systemic non-pulmonary sarcoidosis affecting lacrimal gland&#44; peripheral lymph nodes&#44; parotid gland and the liver&#46;</p></span>"
      ]
      "es" => array:2 [
        "titulo" => "Resumen"
        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">La afectaci&#243;n extrapulmonar aislada en la sarcoidosis es infrecuente y se reporta en el 5 al 9&#37; de las sarcoidosis sist&#233;micas&#44; lo que constituye un desaf&#237;o cl&#237;nico debido a su extenso diagn&#243;stico diferencial&#46; La sarcoidosis extrapulmonar que afecta a m&#225;s de tres &#243;rganos es raramente reportada y hay pocos datos publicados sobre el diagn&#243;stico&#44; la evoluci&#243;n cl&#237;nica y el manejo de estos casos&#46; Presentamos el caso de una mujer de 41 a&#241;os con sarcoidosis sist&#233;mica no pulmonar que afecta a la gl&#225;ndula lacrimal&#44; los ganglios linf&#225;ticos perif&#233;ricos&#44; la gl&#225;ndula par&#243;tida y el h&#237;gado&#46;</p></span>"
      ]
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        "etiqueta" => "Fig&#46; 1"
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        "descripcion" => array:1 [
          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Lacrimal gland enlargement &#40;A&#44; arrow&#41;&#46; The lacrimal gland&#44; supraclavicular lymph node and liver slides stained with haematoxylin and eosin showed confluent epithelioid granulomas without necrosis &#40;A&#8211;C&#42;&#41;&#46; Note the strong histiocytic expression of CD68 in &#40;C&#41;&#46;</p>"
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      "titulo" => "References"
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            0 => array:3 [
              "identificador" => "bib0040"
              "etiqueta" => "1"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Sarcoidosis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:3 [
                            0 => "M&#46;C&#46; Iannuzzi"
                            1 => "B&#46;A&#46; Rybicki"
                            2 => "A&#46;S&#46; Teirstein"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
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                      "Revista" => array:7 [
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                        "paginaInicial" => "2153"
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                        "link" => array:1 [
                          0 => array:2 [
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                            "web" => "Medline"
                          ]
                        ]
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                          "pii" => "S0165178118307613"
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                          "issn" => "01651781"
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                  ]
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              ]
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            1 => array:3 [
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              "etiqueta" => "2"
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                  "contribucion" => array:1 [
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
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                            1 => "Y&#46;C&#46; Cozier"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1177/2040622318790197"
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              ]
            ]
            2 => array:3 [
              "identificador" => "bib0050"
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                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Clinical features of extrapulmonary sarcoidosis without lung involvement"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "W&#46;E&#46; James"
                            1 => "E&#46; Koutroumpakis"
                            2 => "B&#46; Saha"
                            3 => "A&#46; Nathani"
                            4 => "L&#46; Saavedra"
                            5 => "R&#46;M&#46; Yucel"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1016/j.chest.2018.02.003"
                      "Revista" => array:6 [
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                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
            3 => array:3 [
              "identificador" => "bib0055"
              "etiqueta" => "4"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Influence of gender on epidemiology and clinical manifestations of sarcoidosis&#58; a population-based retrospective cohort study 1976&#8211;2013"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:3 [
                            0 => "P&#46; Ungprasert"
                            1 => "C&#46;S&#46; Crowson"
                            2 => "E&#46;L&#46; Matteson"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1007/s00408-016-9952-6"
                      "Revista" => array:6 [
                        "tituloSerie" => "Lung"
                        "fecha" => "2017"
                        "volumen" => "195"
                        "paginaInicial" => "87"
                        "paginaFinal" => "91"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/27709291"
                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
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            4 => array:3 [
              "identificador" => "bib0060"
              "etiqueta" => "5"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "The organ-specific extrapulmonary presentation of sarcoidosis&#58; a frequent occurrence but a challenge to an early diagnosis&#46; A 3-year-long prospective observational study"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:4 [
                            0 => "G&#46; Rizzato"
                            1 => "G&#46; Palmieri"
                            2 => "A&#46;M&#46; Agrati"
                            3 => "C&#46; Zanussi"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:6 [
                        "tituloSerie" => "Sarcoidosis Vasc Diffuse Lung Dis"
                        "fecha" => "2004"
                        "volumen" => "21"
                        "paginaInicial" => "119"
                        "paginaFinal" => "126"
                        "link" => array:1 [
                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/15281433"
                            "web" => "Medline"
                          ]
                        ]
                      ]
                    ]
                  ]
                ]
              ]
            ]
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              "identificador" => "bib0065"
              "etiqueta" => "6"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Natural history of hepatic sarcoidosis and its response to treatment"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
                          "autores" => array:6 [
                            0 => "P&#46;T&#46; Kennedy"
                            1 => "N&#46; Zakaria"
                            2 => "S&#46;B&#46; Modawi"
                            3 => "A&#46;M&#46; Papadopoulou"
                            4 => "I&#46; Murray-Lyon"
                            5 => "R&#46;M&#46; du Bois"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1097/01.meg.0000223911.85739.38"
                      "Revista" => array:6 [
                        "tituloSerie" => "Eur J Gastroenterol Hepatol"
                        "fecha" => "2006"
                        "volumen" => "18"
                        "paginaInicial" => "721"
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                          0 => array:2 [
                            "url" => "https://www.ncbi.nlm.nih.gov/pubmed/16772828"
                            "web" => "Medline"
                          ]
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                      ]
                    ]
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                ]
              ]
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              "identificador" => "bib0070"
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              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Extrapulmonary sarcoidosis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:1 [
                            0 => "M&#46;A&#46; Judson"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
                      "Revista" => array:5 [
                        "tituloSerie" => "Semin Respir Crit Care Med"
                        "fecha" => "2007"
                        "volumen" => "28"
                        "paginaInicial" => "83"
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                      ]
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        "identificador" => "xack602268"
        "titulo" => "Acknowledgements"
        "texto" => "<p id="par0060" class="elsevierStylePara elsevierViewall">We are thankful to Dr&#46; Naranjo from de Department of Anatomical Pathology for their help in the preparation of the histological sections for the presented case&#46; Also&#44; we thank to Juan Vilches for assistance in technique support&#46;</p>"
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      ]
    ]
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