What is your diagnosis?
Case for diagnosis. Ulcerated lesions: a diagnostic challenge in Behçet's syndrome
Lissiê Lunardi Sbroglio
, Karin Milleni de Araújo, Bianca Passos Leite dos Santos, Alexandre Carlos Gripp
Corresponding author
Department of Dermatology, Hospital Universitário Pedro Ernesto, Universidade do Estado do Rio de Janeiro, Rio de Janeiro, RJ, Brazil
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Ulcerated lesions: a diagnostic challenge in Behçet's syndrome" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "494" "paginaFinal" => "496" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Lissiê Lunardi Sbroglio, Karin Milleni de Araújo, Bianca Passos Leite dos Santos, Alexandre Carlos Gripp" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Lissiê Lunardi" "apellidos" => "Sbroglio" "email" => array:1 [ 0 => "lissiesbroglio@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Karin Milleni de" "apellidos" => "Araújo" ] 2 => array:2 [ "nombre" => "Bianca Passos Leite dos" "apellidos" => "Santos" ] 3 => array:2 [ "nombre" => "Alexandre Carlos" "apellidos" => "Gripp" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Hospital Universitário Pedro Ernesto, Universidade do Estado do Rio de Janeiro, Rio de Janeiro, RJ, Brazil" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 443 "Ancho" => 905 "Tamanyo" => 53500 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Patient with progressive improvement of the condition after starting drug treatment (July 2019).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case report</span><p id="par0005" class="elsevierStylePara elsevierViewall">This is a case report of a male patient, 42 years old, with ulcerated lesions on the lower limbs for 12 months. He had been treated in the primary health care services as a case of recurrent cellulitis. Due to therapeutic failure, after cycles of antibiotics and low doses of corticosteroids, he was referred for evaluation at the authors’ Dermatology Unit. The initial clinical examination revealed papulopustular, purpuric and ulcerative necrotic lesions predominantly located on the lower limbs (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The diagnostic hypotheses were Behçet's syndrome (BS), granulomatosis with polyangiitis, eosinophilic granulomatosis with polyangiitis, and endocarditis. Complementary exams (autoantibody screening, investigation for infectious diseases, echocardiography, electroneuromyography of the lower limbs, chest and sinuses computed tomography and fundoscopy) showed no alterations. The skin biopsy was non specific (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and the pathergy test was positive (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The HLA-B typing test showed the presence of allele 51. During the follow-up, he had acne-like lesions on the face and painful ulcers on the scrotum, with no lesions in the oral mucosa (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">What is your diagnosis?</span><p id="par0015" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">a)</span><p id="par0020" class="elsevierStylePara elsevierViewall">Granulomatosis with polyangiitis</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">b)</span><p id="par0025" class="elsevierStylePara elsevierViewall">Endocarditis</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">c)</span><p id="par0030" class="elsevierStylePara elsevierViewall">Behçet's syndrome</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">d)</span><p id="par0035" class="elsevierStylePara elsevierViewall">eosinophilic granulomatosis with polyangiitis</p></li></ul></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0040" class="elsevierStylePara elsevierViewall">Based on the most recent international criteria, the diagnosis of BS was established.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Treatment with colchicine 1.5 mg/day associated with prednisone 0.5 mg/kg/day was started, with disease improvement (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>). The patient is undergoing follow-up by a multidisciplinary team, with no systemic involvement to date. BS is a multisystemic vasculitis with acute inflammatory outbreaks that affect vessels of any caliber.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> The multifactorial etiopathogenesis suggests environmental agents (differences in the manifestation pattern in endemic and non-endemic areas), complex genetic factors, mainly associated with the HLA-B51 – MHC class I allele, which is important in neutrophil activation.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">The initial and most common characteristics are recurrent oral and genital mucosal ulcerations, which are commonly painful and multiple, with well-defined edges, resolving with punched out scars. Pustular, purpuric, ulcerated lesions, erythema nodosum-like, and migratory thrombophlebitis have been described.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The concept of BS phenotyping is used, with four main phenotypes: skin-mucosal involvement, skin, mucosal and joint involvement, vascular disease, and ocular involvement.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> There is no clinical finding or complementary examination which is pathognomonic.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The most recent international diagnostic criteria do not include oral ulceration as mandatory.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Misdiagnoses have been associated with insufficient collection of semiological data and the tendency that some professionals have towards a greater appreciation of technology (laboratory and imaging tests). In this case, no specific complementary exam contributed to the diagnosis, since it is entirely based on clinical criteria, with the exception of the pathergy test.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The treatment aims at symptom remission, not being curative.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Topical agents (corticosteroids, calcineurin inhibitors and antibiotics) are used for mucocutaneous lesions; for extensive/refractory lesions or those associated with musculoskeletal conditions, colchicine, systemic corticosteroids, non-steroidal anti-inflammatory drugs, and dapsone are used; for eye symptoms, azathioprine, cyclosporine, mycophenolate mofetil, thalidomide, and methotrexate are used and for systemic involvement, pulse therapy with corticosteroids and cyclophosphamide and anti-TNF-alpha are the treatment of choice. The following treatments are under study: phosphodiesterase-4 inhibitor, interleukin-1 receptor antagonist, IL-6 and IL-12/23 inhibitors.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7,8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the case reported here, despite the exuberance of the cutaneous lesions, there were no lesions on the oral mucosa, which may have influenced the delay in the diagnosis. Considering BS in the presence of ulcerated lesions in the lower limbs, especially if associated with genital and/or oral lesions, is crucial for specific conduct to be adopted, reducing unnecessary treatments and the negative impact on the patients’ quality of life.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors’ contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Lissiê Lunardi Sbroglio: Design and planning of the study; drafting and editing of the manuscript; critical review of the literature.</p><p id="par0075" class="elsevierStylePara elsevierViewall">Karin Milleni de Araújo: Critical review of the literature; critical review of the manuscript.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Bianca Passos Leite dos Santos: Critical review of the literature; intellectual participation in propaedeutic and/or therapeutic conduct of the studied case.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Alexandre Carlos Gripp: Approval of the final version of the manuscript; critical review of the manuscript.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0090" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1550374" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1400161" "titulo" => "Keywords" ] 2 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 3 => array:2 [ "identificador" => "sec0010" "titulo" => "What is your diagnosis?" ] 4 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 5 => array:2 [ "identificador" => "sec0020" "titulo" => "Financial support" ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Authors’ contributions" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflicts of interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-08-25" "fechaAceptado" => "2021-01-13" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1400161" "palabras" => array:4 [ 0 => "Behçet syndrome" 1 => "Delayed diagnosis" 2 => "Diagnosis, differential" 3 => "Signs and symptoms" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">This study reports the clinical case of a 42-year-old patient with ulcerated lesions who was followed up by general practitioners with the diagnosis of recurrent cellulitis. However, when referred to the Dermatology division a diagnosis of Behçet's syndrome was established based on clinical criteria. Although there are defined clinical criteria for this syndrome, sometimes its diagnosis can be challenging, due to lack of knowledge of the disease and extremely heterogeneous clinical phenotype. The authors highlight the potential difficulties in establishing the diagnosis considering the multiple clinical findings during the investigation process, contributing to the risk of increased morbidity and mortality.</p></span>" ] ] "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Sbroglio LL, Araújo KM, Santos BPL, Gripp AC. Case for diagnosis. Ulcerated lesions: a diagnostic challenge in Behçet's syndrome. An Bras Dermatol. 2021;96:494–6.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital Universitário Pedro Ernesto, Rio de Janeiro, RJ, Brazil.</p>" ] ] "multimedia" => array:4 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 548 "Ancho" => 905 "Tamanyo" => 71593 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Purpuric lesions on the hands; papulopustular, purpuric, and ulceronecrotic lesions on the lower limbs, and ulceronecrotic lesions on the gluteal region and elbow.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 715 "Ancho" => 905 "Tamanyo" => 64922 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A), (Left) Anatomopathological analysis showing diffuse cicatricial fibrosis and vascular proliferation (Hematoxylin & eosin, ×40). (Right) Anatomopathological analysis showing perivascular inflammation with a predominance of neutrophils (Hematoxylin & eosin, ×200). (B), (Left) Pathergy test with positive reading (right).</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 762 "Ancho" => 905 "Tamanyo" => 113906 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Scrotal ulcers and acne-like lesions on the malar regions.</p>" ] ] 3 => array:8 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 443 "Ancho" => 905 "Tamanyo" => 53500 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Patient with progressive improvement of the condition after starting drug treatment (July 2019).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Phenotypes in Behçet’s syndrome" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "E. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 12 | 6 | 18 |
| 2024 October | 106 | 63 | 169 |
| 2024 September | 114 | 82 | 196 |
| 2024 August | 150 | 112 | 262 |
| 2024 July | 133 | 102 | 235 |
| 2024 June | 114 | 76 | 190 |
| 2024 May | 139 | 58 | 197 |
| 2024 April | 127 | 81 | 208 |
| 2024 March | 89 | 59 | 148 |
| 2024 February | 92 | 73 | 165 |
| 2024 January | 72 | 41 | 113 |
| 2023 December | 67 | 64 | 131 |
| 2023 November | 88 | 83 | 171 |
| 2023 October | 65 | 71 | 136 |
| 2023 September | 66 | 76 | 142 |
| 2023 August | 58 | 34 | 92 |
| 2023 July | 101 | 26 | 127 |
| 2023 June | 97 | 34 | 131 |
| 2023 May | 122 | 18 | 140 |
| 2023 April | 96 | 14 | 110 |
| 2023 March | 104 | 36 | 140 |
| 2023 February | 53 | 39 | 92 |
| 2023 January | 46 | 34 | 80 |
| 2022 December | 50 | 35 | 85 |
| 2022 November | 68 | 53 | 121 |
| 2022 October | 105 | 50 | 155 |
| 2022 September | 50 | 65 | 115 |
| 2022 August | 51 | 58 | 109 |
| 2022 July | 56 | 61 | 117 |
| 2022 June | 51 | 53 | 104 |
| 2022 May | 41 | 67 | 108 |
| 2022 April | 65 | 52 | 117 |
| 2022 March | 58 | 77 | 135 |
| 2022 February | 47 | 42 | 89 |
| 2022 January | 85 | 88 | 173 |
| 2021 December | 51 | 72 | 123 |
| 2021 November | 43 | 59 | 102 |
| 2021 October | 69 | 108 | 177 |
| 2021 September | 66 | 87 | 153 |
| 2021 August | 93 | 89 | 182 |
| 2021 July | 125 | 64 | 189 |
| 2021 June | 63 | 58 | 121 |
| 2021 May | 14 | 22 | 36 |
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