Case Report
Behçet's Disease With Intestinal Perforation: A Case Report
Enfermedad de Behçet con perforación intestinal: a propósito de un caso
Maricel Della Maggiora
, Andrea Baños, Virginia Paolini, Dario Florio, Lorena Takashima, Pedro Aicardi
Corresponding author
Servicio de Reumatología, APEsa, Buenos Aires, Argentina
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Thus, whether it should be considered a vasculitis is a matter of debate.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case Report</span><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 34-year-old Caucasian who had been hospitalized with a syndrome that had begun 21 days earlier. It consisted of high fever of unknown origin, together with weakness and a nodular eruption that left scars in both upper and lower limbs, and no ophthalmologic manifestations.</p><p id="par0015" class="elsevierStylePara elsevierViewall">He reported an 8-year history of intermittent painless oral and genital ulcers that appeared and disappeared spontaneously, with no definite diagnosis.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Laboratory tests showed: erythrocyte sedimentation rate >120<span class="elsevierStyleHsp" style=""></span>mm/h, C-reactive protein 400<span class="elsevierStyleHsp" style=""></span>mg/dL and negative results on immunological tests (antiphospholipid antibodies, antineutrophil cytoplasmic antibodies, anticardiolipin antibodies and the venereal disease research laboratory test). Antigen tests, viral serology and a test for human leukocyte antigen (HLA)-B27 were negative. An electrophoretic protein profile was normal. Immunoglobulin (Ig) A level was 443<span class="elsevierStyleHsp" style=""></span>mg/dL. Parasitological analysis of stool was negative.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Thoracic and abdominal computed tomography revealed bilateral pleural effusion, pneumoperitoneum, peritoneal membrane thickening, free fluid in the peritoneal cavity and enlarged lymph nodes. Therefore, he was scheduled for surgery, and was found to have numerous intestinal perforations (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). It was necessary to perform subtotal colectomy and ileostomy. Treatment with meprednisone at 40<span class="elsevierStyleHsp" style=""></span>mg/day resulted in a favorable clinical course. The dose was reduced to 8<span class="elsevierStyleHsp" style=""></span>mg/day, accompanied by azathioprine at 100<span class="elsevierStyleHsp" style=""></span>mg/day, which resulted in 1 year without relapses. A video recording of a follow-up colonoscopy showed active lesions, and he began to receive 40<span class="elsevierStyleHsp" style=""></span>mg of subcutaneous adalimumab every 15 days,<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">1–3</span></a> which achieved clinical and histopathological remission after 4 months of treatment.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a></p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Ancillary histopathological studies of a tissue specimen demonstrated colonic ulceration with perilesional inflammation; a skin biopsy revealed septal lobular panniculitis with mononuclear and polymorphonuclear cell infiltrate; and nonspecific inflammation was observed in the scrotal ulcer. Testing was positive for HLA-B52<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> and negative for HLA-B51. The pathergy test was negative.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conclusions</span><p id="par0035" class="elsevierStylePara elsevierViewall">Patients with Behçet's disease can experience oral and genital ulcers for years, with no definitive diagnosis, and consult their physicians only when serious complications arise. Since the diagnosis is mainly clinical, primary care physicians should be familiar with the characteristics of this disease,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> especially in countries like Argentina in which the prevalence is low and the evidence is concealed in a variety of symptoms that must be associated to discovery it. Therefore any delay in the diagnosis increases the rates of mortality and morbidity affecting patients with BD.</p><p id="par0040" class="elsevierStylePara elsevierViewall">To date, when gastrointestinal involvement is detected, good responses have been achieved with sulfasalazine, azathioprine and corticosteroids at low or high doses or as pulse therapy, as well as with pulse cyclophosphamide. However, in the case of intestinal perforation, the treatment must be aggressive and can be initiated directly with adalimumab.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Ethical Disclosures</span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Protection of human and animal subjects</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Confidentiality of data</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Right to privacy and informed consent</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that no patient data appear in this article.</p></span></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Conflicts of Interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres835739" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec831487" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres835740" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec831488" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case Report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Conclusions" ] 7 => array:3 [ "identificador" => "sec0020" "titulo" => "Ethical Disclosures" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0025" "titulo" => "Protection of human and animal subjects" ] 1 => array:2 [ "identificador" => "sec0030" "titulo" => "Confidentiality of data" ] 2 => array:2 [ "identificador" => "sec0035" "titulo" => "Right to privacy and informed consent" ] ] ] 8 => array:2 [ "identificador" => "sec0040" "titulo" => "Conflicts of Interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2016-01-29" "fechaAceptado" => "2016-03-16" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec831487" "palabras" => array:3 [ 0 => "Behçet's disease" 1 => "Intestinal complications" 2 => "Adalimumab" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec831488" "palabras" => array:3 [ 0 => "Enfermedad de Behçet" 1 => "Complicaciones intestinales" 2 => "Adalimumab" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 34-year-old man with an 8-year history of outbreaks of oral and genital ulcers presented with high fever and skin lesions that had developed 21 days earlier, without abdominal pain. Computed tomography revealed intestinal perforation requiring surgery. After treatment with corticosteroids and azathioprine proved to be ineffective, it was switched to adalimumab.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Paciente de sexo masculino de 34 años con antecedente de úlceras orales y genitales que cursaban en brotes de 8 años de evolución, se presenta con fiebre elevada y lesiones en piel durante 21 días, sin dolor abdominal. Se realiza tomografía computarizada, observándose perforación intestinal, por lo que es intervenido quirúrgicamente. Es tratado con corticoides y azatioprina. Luego, por falta de respuesta, se le administra adalimumab.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Della Maggiora M, Baños A, Paolini V, Florio D, Takashima L, Aicardi P. Enfermedad de Behçet con perforación intestinal: a propósito de un caso. Reumatol Clin. 2017;13:171–172.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 572 "Ancho" => 900 "Tamanyo" => 85970 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Image of intestinal perforation in a patient with Behçet's disease.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "New evidence-based treatment approach in Behcet's disease" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "E. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 15 | 26 |
| 2024 October | 92 | 26 | 118 |
| 2024 September | 94 | 13 | 107 |
| 2024 August | 85 | 48 | 133 |
| 2024 July | 82 | 28 | 110 |
| 2024 June | 98 | 38 | 136 |
| 2024 May | 97 | 31 | 128 |
| 2024 April | 98 | 29 | 127 |
| 2024 March | 69 | 28 | 97 |
| 2024 February | 63 | 31 | 94 |
| 2024 January | 50 | 21 | 71 |
| 2023 December | 56 | 29 | 85 |
| 2023 November | 80 | 29 | 109 |
| 2023 October | 59 | 28 | 87 |
| 2023 September | 68 | 39 | 107 |
| 2023 August | 46 | 9 | 55 |
| 2023 July | 74 | 34 | 108 |
| 2023 June | 53 | 23 | 76 |
| 2023 May | 88 | 39 | 127 |
| 2023 April | 40 | 8 | 48 |
| 2023 March | 97 | 37 | 134 |
| 2023 February | 83 | 35 | 118 |
| 2023 January | 140 | 15 | 155 |
| 2022 December | 99 | 46 | 145 |
| 2022 November | 89 | 30 | 119 |
| 2022 October | 99 | 25 | 124 |
| 2022 September | 81 | 34 | 115 |
| 2022 August | 76 | 37 | 113 |
| 2022 July | 76 | 45 | 121 |
| 2022 June | 72 | 30 | 102 |
| 2022 May | 104 | 46 | 150 |
| 2022 April | 90 | 45 | 135 |
| 2022 March | 96 | 50 | 146 |
| 2022 February | 86 | 36 | 122 |
| 2022 January | 81 | 43 | 124 |
| 2021 December | 60 | 45 | 105 |
| 2021 November | 62 | 44 | 106 |
| 2021 October | 84 | 70 | 154 |
| 2021 September | 60 | 42 | 102 |
| 2021 August | 50 | 46 | 96 |
| 2021 July | 47 | 45 | 92 |
| 2021 June | 59 | 37 | 96 |
| 2021 May | 64 | 45 | 109 |
| 2021 April | 131 | 97 | 228 |
| 2021 March | 71 | 34 | 105 |
| 2021 February | 52 | 12 | 64 |
| 2021 January | 49 | 18 | 67 |
| 2020 December | 40 | 24 | 64 |
| 2020 November | 51 | 19 | 70 |
| 2020 October | 41 | 23 | 64 |
| 2020 September | 51 | 45 | 96 |
| 2020 August | 43 | 24 | 67 |
| 2020 July | 34 | 18 | 52 |
| 2020 June | 71 | 33 | 104 |
| 2020 May | 48 | 23 | 71 |
| 2020 April | 51 | 11 | 62 |
| 2020 March | 25 | 7 | 32 |
| 2019 January | 3 | 0 | 3 |
| 2018 May | 6 | 0 | 6 |
| 2018 April | 44 | 6 | 50 |
| 2018 March | 93 | 8 | 101 |
| 2018 February | 24 | 11 | 35 |
| 2018 January | 49 | 7 | 56 |
| 2017 December | 35 | 19 | 54 |
| 2017 November | 28 | 13 | 41 |
| 2017 October | 24 | 8 | 32 |
| 2017 September | 27 | 16 | 43 |
| 2017 August | 40 | 13 | 53 |
| 2017 July | 43 | 17 | 60 |
| 2017 June | 68 | 38 | 106 |
| 2017 May | 62 | 23 | 85 |
| 2017 April | 3 | 20 | 23 |
| 2017 March | 3 | 12 | 15 |
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