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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Chronic recurrent granulomatous processes in the anogenital area present with ulcers&#44; fissures&#44; and lymphedema&#59; histopathology reveals nonnecrotizing granulomatous inflammation&#46; Crohn disease is the most common etiologic factor&#44; but cases in which no underlying cause is evident have been grouped under the term anogenital granulomatosis&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The first case we report is that of a 52-year-old woman with excrescent lesions that had a pseudocondylomatous appearance and fissures in the gluteal cleft that had started 6 months earlier&#46; The lesions were excised but she did not return until 5 years later&#44; when she sought care for chronic recurrent vulvar and perineal lesions&#46; She had ulcers&#44; marked edema of the vulva&#44; longitudinal fissures in the folds&#44; and indurated plaques that were excrescent in the gluteal cleft &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Histopathology of both the vulvar and the perianal areas revealed a lymphocytic infiltrate in the reticular dermis with nonnecrotizing granulomas consisting of multinucleated giant cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41;&#46; Additional tests&#44; including complete blood count&#44; biochemistry&#44; chest radiograph&#44; and cultures yielded no findings&#44; except for an elevated erythrocyte sedimentation rate &#40;ESR&#41; of 52<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46; A colonoscopy with colorectal biopsies ruled out inflammatory bowel disease&#46; The patient was treated with topical corticosteroids&#44; salicylates&#44; and oral corticosteroids&#59; the lesions responded fully to the last treatment but recurred when they were suspended&#46; She was subsequently treated with adalimumab &#40;40<span class="elsevierStyleHsp" style=""></span>mg&#47;15<span class="elsevierStyleHsp" style=""></span>d&#41; but showed no response after 4 months&#46; Fifteen years after the initial episode the patient had not developed systemic symptoms&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second case is that of a 51-year-old woman who presented with painful erosions and vulvar edema dating from 5 months earlier&#46; For 6 years she had also had recurrent perianal suppurative plaques and fissures that had been diagnosed as hidradenitis suppurativa&#46; Physical examination revealed the granulomatous appearance of the vulvar mucosa&#44; erosions on the inside of the labia minora &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#44; and longitudinal fissures in the gluteal cleft&#46; Two vulvar biopsies revealed a dense lymphocytic infiltrate of epithelioid cells and multinucleated giant cells forming granulomas without central necrosis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C and D&#41;&#46; Additional tests &#40;complete blood count&#44; biochemistry&#44; cultures&#44; and chest radiograph&#41; were normal or negative&#44; except for a slightly elevated ESR of 25<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46; Crohn disease was ruled out after colonoscopy with biopsies&#46; The patient had no systemic symptoms during the 18 months of follow-up and responded partially to treatment with oral corticosteroids but showed no response to salicylates or to adalimumab &#40;40<span class="elsevierStyleHsp" style=""></span>mg&#47;15<span class="elsevierStyleHsp" style=""></span>d&#41;&#44; which was therefore suspended after 3 months&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The differential diagnosis of chronic granulomatous diseases in the perineum includes extraintestinal Crohn disease&#44; although this condition is unlikely in the absence of intestinal symptoms or perianal fistulas and with normal colonoscopy&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Other possible differential diagnoses are shown in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#46; In view of the clinical presentation&#44; the first diagnostic steps should be biopsy to obtain a specimen for histology &#40;on the basis of special stains and cultures for fungi&#44; bacteria&#44; and mycobacteria&#41;&#46; Additional tests useful to rule out underlying causes include complete blood count&#44; biochemistry&#44; iron profile&#44; ESR&#44; angiotensin-converting enzyme levels&#44; serology for syphilis&#44; and chest radiograph&#46; Colonoscopy is recommended&#44; even in the absence of digestive symptoms&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Chronic genital granulomatosis without direct communication with the gastrointestinal tract can be observed in metastatic Crohn disease&#46; This condition is the least common cutaneous manifestation of Crohn disease and consists of skin lesions separated from the digestive tract by healthy skin&#46; It usually affects women between the second and fourth decades of life and can appear anywhere&#44; including on the genitals&#44; although the lower limbs are the most frequent location&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Clinical manifestations in the genital region are similar to those observed in our patients&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Lesions are associated with involvement of the colon or rectum<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> but usually do not follow a course that runs parallel to the intestinal disease&#46; Chronic genital granulomatosis is associated with long-standing intestinal Crohn disease in 80&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;6</span></a> When the granulomatous process presents first&#44; intestinal involvement usually develops within 4 months to 2 years&#46; The literature offers at least 5 cases<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> of cutaneous Crohn disease in the absence of previously recognized intestinal disease&#44; which did not appear during follow-up either&#46; Some authors nonetheless recommend reserving this diagnosis for cases in which intestinal involvement has been demonstrated&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Orofacial granulomatosis or cheilitis granulomatosa&#44; considered a monosymptomatic form of Melkersson&#8211;Rosenthal syndrome&#44; shares some of the clinical and histological features of the anogenital granulomatosis in our 2 cases&#46; Cheilitis granulomatosa presents as persistent and recurrent labial swelling&#59; nonnecrotizing granulomas are sometimes associated with ulceration and gingival hyperplasia or cobblestoning&#46; Anogenital granulomatosis has been suggested to be the genital equivalent of cheilitis granulomatosa<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and although the co-occurrence of these 2 conditions in the same patient is rare&#44; it has been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In 10&#37;&#8211;48&#37; of cases cheilitis granulomatosa and intestinal Crohn disease are associated&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The term anogenital granulomatosis<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> was introduced in 2003 to identify these chronic recurrent conditions with characteristic clinical and histopathologic features that may have different causes&#46; This clinical entity is a unifying concept for others used in the literature &#40;chronic hypertrophic vulvitis&#44; vulvitis granulomatosa&#44; chronic edema of the vulva&#44; Melkersson&#8211;Rosenthal vulvitis and anoperineitis granulomatosa&#41; and is especially useful for cases of unknown etiology and those highly suggestive of metastatic Crohn disease in the absence of established intestinal disease&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Therapeutic management of this condition is difficult and there is no set protocol to follow because of the lack of case series and randomized trials&#46; Suggested treatments have obtained mixed and sometimes unsatisfactory results marked by frequent relapse after treatment is discontinued&#46; The reported options include topical&#44; intralesional&#44; and oral corticosteroids&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> salicylates&#44; antibiotics<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> such as metronidazole<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;10</span></a> and ciprofloxacin&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and immunosuppressants such as azathioprine and ciclosporin&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> More recently&#44; anti-tumor necrosis factor monoclonal antibodies such as infliximab and adalimumab have given good results&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">We have reported 2 cases of idiopathic anogenital granulomatosis in which possible underlying causes were ruled out and no associated systemic symptoms developed even after years of follow-up&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara">Please cite this article as&#58; Villar M&#44; Petiti G&#44; Guerra A&#44; Vanaclocha F&#46; Granulomatosis anogenitales&#46; Actas Dermosifiliogr&#46; 2012&#59;103&#58;76&#8211;79&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Vulva&#44; showing numerous ulcers&#44; fissures arranged longitudinally in the folds&#44; and edema of the labia majora&#44; labia minora&#44; and clitoral hood&#46; In the gluteal cleft&#44; excrescent plaques with a pseudocondylomatous appearance and longitudinal cracks&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Lymphocytic infiltrate occupying the reticular dermis&#44; showing granulomatous structures without central necrosis &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;12&#46;5&#41;&#46; &#40;B&#41; Detail of a granuloma composed of multinucleated giant cells and epithelioid cells and surrounded by lymphocytes &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;400&#41;&#46; &#40;C&#41; Dense lymphocytic infiltrate occupying a mucous membrane &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;40&#41;&#46; &#40;D&#41; At higher magnification&#44; epithelioid cells are seen forming nonnecrotizing granulomas &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;400&#41;&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Edematous vulvar mucosa&#44; with a granulomatous appearance&#46; Erosions on the inner surface of the labia minora&#46;</p>"
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                  <table border="0" frame="\n
                  \t\t\t\t\tvoid\n
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                  \t\t\t\t\ttable-head\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t" style="border-bottom: 2px solid black">Noninfectious Causes&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-head\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t" style="border-bottom: 2px solid black">Infectious Causes&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Crohn disease&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Tuberculosis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Sarcoidosis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Lymphogranuloma venereum&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Foreign body granuloma&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Granuloma inguinale&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Hidradenitis suppurativa&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Syphilis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Beh&#231;et disease&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Leprosy&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Pyoderma gangrenosum&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Deep mycoses&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Lymphoproliferative diseases&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="" valign="\n
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                  \t\t\t\t">&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t">Melkersson&#8211;Rosenthal syndrome&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="" valign="\n
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                  \t\t\t\t">&nbsp;\t\t\t\t\t\t\n
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Differential Diagnosis of Anogenital Granulomatosis&#46;</p>"
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      "titulo" => "References"
      "seccion" => array:1 [
        0 => array:2 [
          "identificador" => "bibs0005"
          "bibliografiaReferencia" => array:10 [
            0 => array:3 [
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              "referencia" => array:1 [
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                  "contribucion" => array:1 [
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                      "titulo" => "Ano-genital granulomatosis&#58; the counterpart of oro-facial granulomatosis"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
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                            0 => "M&#46;R&#46; van der Scheur"
                            1 => "R&#46;I&#46; van der Waal"
                            2 => "I&#46; van der Waal"
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                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:1 [
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                        "link" => array:1 [
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                            "web" => "Medline"
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                  ]
                ]
              ]
            ]
            1 => array:3 [
              "identificador" => "bib0010"
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              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:3 [
                            0 => "H&#46; Hackel"
                            1 => "A&#46;A&#46; Hartmann"
                            2 => "G&#46; Burg"
                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
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            2 => array:3 [
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                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => true
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                            0 => "I&#46; Palamaras"
                            1 => "J&#46; El-Jabbour"
                            2 => "N&#46; Pietropaolo"
                            3 => "P&#46; Thomson"
                            4 => "S&#46; Mann"
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                          ]
                        ]
                      ]
                    ]
                  ]
                  "host" => array:1 [
                    0 => array:2 [
                      "doi" => "10.1111/j.1468-3083.2008.02741.x"
                      "Revista" => array:6 [
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                        "volumen" => "22"
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                            "web" => "Medline"
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                    ]
                  ]
                ]
              ]
            ]
            3 => array:3 [
              "identificador" => "bib0020"
              "etiqueta" => "4"
              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Enfermedad de Crohn metast&#225;sica&#58; a prop&#243;sito de un caso con afectaci&#243;n vulvar"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "P&#46; G&#243;mez-Centeno"
                            1 => "L&#46; Casas"
                            2 => "F&#46; Vald&#233;s-Tasc&#243;n"
                            3 => "C&#46; Peteiro"
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                          ]
                        ]
                      ]
                    ]
                  ]
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                  "contribucion" => array:1 [
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                      "autores" => array:1 [
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                          "etal" => false
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                            0 => "S&#46; Bel Pla"
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                            2 => "D&#46; Garc&#237;a Fern&#225;ndez"
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                          ]
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                          "etal" => true
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                            0 => "S&#46; Thiriar"
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                    0 => array:2 [
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                            0 => "T&#46; Bogenrieder"
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            8 => array:3 [
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Case and Research Letters
Anogenital Granulomatosis
Granulomatosis anogenitales
M. Villar
Corresponding author
mariavb20@hotmail.com

Corresponding author.
, G. Petiti, A. Guerra, F. Vanaclocha
Servicio de Dermatología, Hospital Universitario 12 de octubre, Madrid, Spain
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longitudinal fissures in the folds&#44; and indurated plaques that were excrescent in the gluteal cleft &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Histopathology of both the vulvar and the perianal areas revealed a lymphocytic infiltrate in the reticular dermis with nonnecrotizing granulomas consisting of multinucleated giant cells &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A and B&#41;&#46; Additional tests&#44; including complete blood count&#44; biochemistry&#44; chest radiograph&#44; and cultures yielded no findings&#44; except for an elevated erythrocyte sedimentation rate &#40;ESR&#41; of 52<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46; A colonoscopy with colorectal biopsies ruled out inflammatory bowel disease&#46; The patient was treated with topical corticosteroids&#44; salicylates&#44; and oral corticosteroids&#59; the lesions responded fully to the last treatment but recurred when they were suspended&#46; She was subsequently treated with adalimumab &#40;40<span class="elsevierStyleHsp" style=""></span>mg&#47;15<span class="elsevierStyleHsp" style=""></span>d&#41; but showed no response after 4 months&#46; Fifteen years after the initial episode the patient had not developed systemic symptoms&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The second case is that of a 51-year-old woman who presented with painful erosions and vulvar edema dating from 5 months earlier&#46; For 6 years she had also had recurrent perianal suppurative plaques and fissures that had been diagnosed as hidradenitis suppurativa&#46; Physical examination revealed the granulomatous appearance of the vulvar mucosa&#44; erosions on the inside of the labia minora &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#44; and longitudinal fissures in the gluteal cleft&#46; Two vulvar biopsies revealed a dense lymphocytic infiltrate of epithelioid cells and multinucleated giant cells forming granulomas without central necrosis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>C and D&#41;&#46; Additional tests &#40;complete blood count&#44; biochemistry&#44; cultures&#44; and chest radiograph&#41; were normal or negative&#44; except for a slightly elevated ESR of 25<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#46; Crohn disease was ruled out after colonoscopy with biopsies&#46; The patient had no systemic symptoms during the 18 months of follow-up and responded partially to treatment with oral corticosteroids but showed no response to salicylates or to adalimumab &#40;40<span class="elsevierStyleHsp" style=""></span>mg&#47;15<span class="elsevierStyleHsp" style=""></span>d&#41;&#44; which was therefore suspended after 3 months&#46;</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The differential diagnosis of chronic granulomatous diseases in the perineum includes extraintestinal Crohn disease&#44; although this condition is unlikely in the absence of intestinal symptoms or perianal fistulas and with normal colonoscopy&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Other possible differential diagnoses are shown in <a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>&#46; In view of the clinical presentation&#44; the first diagnostic steps should be biopsy to obtain a specimen for histology &#40;on the basis of special stains and cultures for fungi&#44; bacteria&#44; and mycobacteria&#41;&#46; Additional tests useful to rule out underlying causes include complete blood count&#44; biochemistry&#44; iron profile&#44; ESR&#44; angiotensin-converting enzyme levels&#44; serology for syphilis&#44; and chest radiograph&#46; Colonoscopy is recommended&#44; even in the absence of digestive symptoms&#46;</p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Chronic genital granulomatosis without direct communication with the gastrointestinal tract can be observed in metastatic Crohn disease&#46; This condition is the least common cutaneous manifestation of Crohn disease and consists of skin lesions separated from the digestive tract by healthy skin&#46; It usually affects women between the second and fourth decades of life and can appear anywhere&#44; including on the genitals&#44; although the lower limbs are the most frequent location&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Clinical manifestations in the genital region are similar to those observed in our patients&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Lesions are associated with involvement of the colon or rectum<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> but usually do not follow a course that runs parallel to the intestinal disease&#46; Chronic genital granulomatosis is associated with long-standing intestinal Crohn disease in 80&#37; of cases&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;6</span></a> When the granulomatous process presents first&#44; intestinal involvement usually develops within 4 months to 2 years&#46; The literature offers at least 5 cases<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> of cutaneous Crohn disease in the absence of previously recognized intestinal disease&#44; which did not appear during follow-up either&#46; Some authors nonetheless recommend reserving this diagnosis for cases in which intestinal involvement has been demonstrated&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Orofacial granulomatosis or cheilitis granulomatosa&#44; considered a monosymptomatic form of Melkersson&#8211;Rosenthal syndrome&#44; shares some of the clinical and histological features of the anogenital granulomatosis in our 2 cases&#46; Cheilitis granulomatosa presents as persistent and recurrent labial swelling&#59; nonnecrotizing granulomas are sometimes associated with ulceration and gingival hyperplasia or cobblestoning&#46; Anogenital granulomatosis has been suggested to be the genital equivalent of cheilitis granulomatosa<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and although the co-occurrence of these 2 conditions in the same patient is rare&#44; it has been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> In 10&#37;&#8211;48&#37; of cases cheilitis granulomatosa and intestinal Crohn disease are associated&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The term anogenital granulomatosis<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> was introduced in 2003 to identify these chronic recurrent conditions with characteristic clinical and histopathologic features that may have different causes&#46; This clinical entity is a unifying concept for others used in the literature &#40;chronic hypertrophic vulvitis&#44; vulvitis granulomatosa&#44; chronic edema of the vulva&#44; Melkersson&#8211;Rosenthal vulvitis and anoperineitis granulomatosa&#41; and is especially useful for cases of unknown etiology and those highly suggestive of metastatic Crohn disease in the absence of established intestinal disease&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Therapeutic management of this condition is difficult and there is no set protocol to follow because of the lack of case series and randomized trials&#46; Suggested treatments have obtained mixed and sometimes unsatisfactory results marked by frequent relapse after treatment is discontinued&#46; The reported options include topical&#44; intralesional&#44; and oral corticosteroids&#44;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> salicylates&#44; antibiotics<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> such as metronidazole<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;10</span></a> and ciprofloxacin&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and immunosuppressants such as azathioprine and ciclosporin&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> More recently&#44; anti-tumor necrosis factor monoclonal antibodies such as infliximab and adalimumab have given good results&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">We have reported 2 cases of idiopathic anogenital granulomatosis in which possible underlying causes were ruled out and no associated systemic symptoms developed even after years of follow-up&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara">Please cite this article as&#58; Villar M&#44; Petiti G&#44; Guerra A&#44; Vanaclocha F&#46; Granulomatosis anogenitales&#46; Actas Dermosifiliogr&#46; 2012&#59;103&#58;76&#8211;79&#46;</p>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Vulva&#44; showing numerous ulcers&#44; fissures arranged longitudinally in the folds&#44; and edema of the labia majora&#44; labia minora&#44; and clitoral hood&#46; In the gluteal cleft&#44; excrescent plaques with a pseudocondylomatous appearance and longitudinal cracks&#46;</p>"
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          "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Lymphocytic infiltrate occupying the reticular dermis&#44; showing granulomatous structures without central necrosis &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;12&#46;5&#41;&#46; &#40;B&#41; Detail of a granuloma composed of multinucleated giant cells and epithelioid cells and surrounded by lymphocytes &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;400&#41;&#46; &#40;C&#41; Dense lymphocytic infiltrate occupying a mucous membrane &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;40&#41;&#46; &#40;D&#41; At higher magnification&#44; epithelioid cells are seen forming nonnecrotizing granulomas &#40;hematoxylin&#8211;eosin&#44; original magnification &#215;400&#41;&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Edematous vulvar mucosa&#44; with a granulomatous appearance&#46; Erosions on the inner surface of the labia minora&#46;</p>"
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                  \t\t\t\t\ttable-head\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t" style="border-bottom: 2px solid black">Noninfectious Causes&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t" style="border-bottom: 2px solid black">Infectious Causes&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr></thead><tbody title="tbody"><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Crohn disease&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Tuberculosis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Sarcoidosis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Lymphogranuloma venereum&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Foreign body granuloma&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Granuloma inguinale&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Hidradenitis suppurativa&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Syphilis&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Beh&#231;et disease&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Leprosy&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Pyoderma gangrenosum&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">Deep mycoses&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td></tr><tr title="table-row"><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Lymphoproliferative diseases&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="" valign="\n
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                  \t\t\t\t">&nbsp;\t\t\t\t\t\t\n
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                  \t\t\t\t  " align="left" valign="\n
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                  \t\t\t\t">Melkersson&#8211;Rosenthal syndrome&nbsp;\t\t\t\t\t\t\n
                  \t\t\t\t</td><td class="td" title="\n
                  \t\t\t\t\ttable-entry\n
                  \t\t\t\t  " align="" valign="\n
                  \t\t\t\t\ttop\n
                  \t\t\t\t">&nbsp;\t\t\t\t\t\t\n
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          "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Differential Diagnosis of Anogenital Granulomatosis&#46;</p>"
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            1 => array:3 [
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                0 => array:2 [
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                        0 => array:2 [
                          "etal" => false
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                            0 => "H&#46; Hackel"
                            1 => "A&#46;A&#46; Hartmann"
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                          ]
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                        0 => array:2 [
                          "etal" => true
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                            1 => "J&#46; El-Jabbour"
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                  "host" => array:1 [
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            3 => array:3 [
              "identificador" => "bib0020"
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              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Enfermedad de Crohn metast&#225;sica&#58; a prop&#243;sito de un caso con afectaci&#243;n vulvar"
                      "autores" => array:1 [
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                          "etal" => false
                          "autores" => array:5 [
                            0 => "P&#46; G&#243;mez-Centeno"
                            1 => "L&#46; Casas"
                            2 => "F&#46; Vald&#233;s-Tasc&#243;n"
                            3 => "C&#46; Peteiro"
                            4 => "J&#46; Toribio"
                          ]
                        ]
                      ]
                    ]
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              "referencia" => array:1 [
                0 => array:2 [
                  "contribucion" => array:1 [
                    0 => array:2 [
                      "titulo" => "Linfedema vulvar&#58; una manifestaci&#243;n infrecuente de la enfermedad de Crohn metast&#225;sica"
                      "autores" => array:1 [
                        0 => array:2 [
                          "etal" => false
                          "autores" => array:5 [
                            0 => "S&#46; Bel Pla"
                            1 => "V&#46; Garc&#237;a-Patos Briones"
                            2 => "D&#46; Garc&#237;a Fern&#225;ndez"
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                          ]
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                      ]
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Article information
ISSN: 15782190
Original language: English
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