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the diagnosis of BS was established&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Treatment with colchicine 1&#46;5&#160;mg&#47;day associated with prednisone 0&#46;5&#160;mg&#47;kg&#47;day was started&#44; with disease improvement &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; The patient is undergoing follow-up by a multidisciplinary team&#44; with no systemic involvement to date&#46; BS is a multisystemic vasculitis with acute inflammatory outbreaks that affect vessels of any caliber&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> The multifactorial etiopathogenesis suggests environmental agents &#40;differences in the manifestation pattern in endemic and non-endemic areas&#41;&#44; complex genetic factors&#44; mainly associated with the HLA-B51 &#8211; MHC class I allele&#44; which is important in neutrophil activation&#46;<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&#44; which are commonly painful and multiple&#44; with well-defined edges&#44; resolving with punched out scars&#46; Pustular&#44; purpuric&#44; ulcerated lesions&#44; erythema nodosum-like&#44; and migratory thrombophlebitis have been described&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The concept of BS phenotyping is used&#44; with four main phenotypes&#58; skin-mucosal involvement&#44; skin&#44; mucosal and joint involvement&#44; vascular disease&#44; and ocular involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> There is no clinical finding or complementary examination which is pathognomonic&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The most recent international diagnostic criteria do not include oral ulceration as mandatory&#46;<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 &#40;laboratory and imaging tests&#41;&#46; In this case&#44; no specific complementary exam contributed to the diagnosis&#44; since it is entirely based on clinical criteria&#44; with the exception of the pathergy test&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The treatment aims at symptom remission&#44; not being curative&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Topical agents &#40;corticosteroids&#44; calcineurin inhibitors and antibiotics&#41; are used for mucocutaneous lesions&#59; for extensive&#47;refractory lesions or those associated with musculoskeletal conditions&#44; colchicine&#44; systemic corticosteroids&#44; non-steroidal anti-inflammatory drugs&#44; and dapsone are used&#59; for eye symptoms&#44; azathioprine&#44; cyclosporine&#44; mycophenolate mofetil&#44; thalidomide&#44; and methotrexate are used and for systemic involvement&#44; pulse therapy with corticosteroids and cyclophosphamide and anti-TNF-alpha are the treatment of choice&#46; The following treatments are under study&#58; phosphodiesterase-4 inhibitor&#44; interleukin-1 receptor antagonist&#44; IL-6 and IL-12&#47;23 inhibitors&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the case reported here&#44; despite the exuberance of the cutaneous lesions&#44; there were no lesions on the oral mucosa&#44; which may have influenced the delay in the diagnosis&#46; Considering BS in the presence of ulcerated lesions in the lower limbs&#44; especially if associated with genital and&#47;or oral lesions&#44; is crucial for specific conduct to be adopted&#44; reducing unnecessary treatments and the negative impact on the patients&#8217; quality of life&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors&#8217; contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Lissi&#234; Lunardi Sbroglio&#58; Design and planning of the study&#59; drafting and editing of the manuscript&#59; critical review of the literature&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Karin Milleni de Ara&#250;jo&#58; Critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Bianca Passos Leite dos Santos&#58; Critical review of the literature&#59; intellectual participation in propaedeutic and&#47;or therapeutic conduct of the studied case&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">Alexandre Carlos Gripp&#58; Approval of the final version of the manuscript&#59; critical review of the manuscript&#46;</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&#46;</p></span></span>"
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What is your diagnosis?
Case for diagnosis. Ulcerated lesions: a diagnostic challenge in Behçet's syndrome
Lissiê Lunardi Sbroglio
Autor para correspondência
lissiesbroglio@gmail.com

Corresponding author.
, Karin Milleni de Araújo, Bianca Passos Leite dos Santos, Alexandre Carlos Gripp
Department of Dermatology, Hospital Universitário Pedro Ernesto, Universidade do Estado do Rio de Janeiro, Rio de Janeiro, RJ, Brazil
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    "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&#44; 42 years old&#44; with ulcerated lesions on the lower limbs for 12 months&#46; He had been treated in the primary health care services as a case of recurrent cellulitis&#46; Due to therapeutic failure&#44; after cycles of antibiotics and low doses of corticosteroids&#44; he was referred for evaluation at the authors&#8217; Dermatology Unit&#46; The initial clinical examination revealed papulopustular&#44; purpuric and ulcerative necrotic lesions predominantly located on the lower limbs &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The diagnostic hypotheses were Beh&#231;et&#39;s syndrome &#40;BS&#41;&#44; granulomatosis with polyangiitis&#44; eosinophilic granulomatosis with polyangiitis&#44; and endocarditis&#46; Complementary exams &#40;autoantibody screening&#44; investigation for infectious diseases&#44; echocardiography&#44; electroneuromyography of the lower limbs&#44; chest and sinuses computed tomography and fundoscopy&#41; showed no alterations&#46; The skin biopsy was non specific &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; and the pathergy test was positive &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The HLA-B typing test showed the presence of allele 51&#46; During the follow-up&#44; he had acne-like lesions on the face and painful ulcers on the scrotum&#44; with no lesions in the oral mucosa &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</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&#63;</span><p id="par0015" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">a&#41;</span><p id="par0020" class="elsevierStylePara elsevierViewall">Granulomatosis with polyangiitis</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">b&#41;</span><p id="par0025" class="elsevierStylePara elsevierViewall">Endocarditis</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">c&#41;</span><p id="par0030" class="elsevierStylePara elsevierViewall">Beh&#231;et&#39;s syndrome</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">d&#41;</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&#44; the diagnosis of BS was established&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Treatment with colchicine 1&#46;5&#160;mg&#47;day associated with prednisone 0&#46;5&#160;mg&#47;kg&#47;day was started&#44; with disease improvement &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46; The patient is undergoing follow-up by a multidisciplinary team&#44; with no systemic involvement to date&#46; BS is a multisystemic vasculitis with acute inflammatory outbreaks that affect vessels of any caliber&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> The multifactorial etiopathogenesis suggests environmental agents &#40;differences in the manifestation pattern in endemic and non-endemic areas&#41;&#44; complex genetic factors&#44; mainly associated with the HLA-B51 &#8211; MHC class I allele&#44; which is important in neutrophil activation&#46;<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&#44; which are commonly painful and multiple&#44; with well-defined edges&#44; resolving with punched out scars&#46; Pustular&#44; purpuric&#44; ulcerated lesions&#44; erythema nodosum-like&#44; and migratory thrombophlebitis have been described&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The concept of BS phenotyping is used&#44; with four main phenotypes&#58; skin-mucosal involvement&#44; skin&#44; mucosal and joint involvement&#44; vascular disease&#44; and ocular involvement&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> There is no clinical finding or complementary examination which is pathognomonic&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The most recent international diagnostic criteria do not include oral ulceration as mandatory&#46;<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 &#40;laboratory and imaging tests&#41;&#46; In this case&#44; no specific complementary exam contributed to the diagnosis&#44; since it is entirely based on clinical criteria&#44; with the exception of the pathergy test&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The treatment aims at symptom remission&#44; not being curative&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Topical agents &#40;corticosteroids&#44; calcineurin inhibitors and antibiotics&#41; are used for mucocutaneous lesions&#59; for extensive&#47;refractory lesions or those associated with musculoskeletal conditions&#44; colchicine&#44; systemic corticosteroids&#44; non-steroidal anti-inflammatory drugs&#44; and dapsone are used&#59; for eye symptoms&#44; azathioprine&#44; cyclosporine&#44; mycophenolate mofetil&#44; thalidomide&#44; and methotrexate are used and for systemic involvement&#44; pulse therapy with corticosteroids and cyclophosphamide and anti-TNF-alpha are the treatment of choice&#46; The following treatments are under study&#58; phosphodiesterase-4 inhibitor&#44; interleukin-1 receptor antagonist&#44; IL-6 and IL-12&#47;23 inhibitors&#46;<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7&#44;8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the case reported here&#44; despite the exuberance of the cutaneous lesions&#44; there were no lesions on the oral mucosa&#44; which may have influenced the delay in the diagnosis&#46; Considering BS in the presence of ulcerated lesions in the lower limbs&#44; especially if associated with genital and&#47;or oral lesions&#44; is crucial for specific conduct to be adopted&#44; reducing unnecessary treatments and the negative impact on the patients&#8217; quality of life&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors&#8217; contributions</span><p id="par0070" class="elsevierStylePara elsevierViewall">Lissi&#234; Lunardi Sbroglio&#58; Design and planning of the study&#59; drafting and editing of the manuscript&#59; critical review of the literature&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">Karin Milleni de Ara&#250;jo&#58; Critical review of the literature&#59; critical review of the manuscript&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">Bianca Passos Leite dos Santos&#58; Critical review of the literature&#59; intellectual participation in propaedeutic and&#47;or therapeutic conduct of the studied case&#46;</p><p id="par0085" class="elsevierStylePara elsevierViewall">Alexandre Carlos Gripp&#58; Approval of the final version of the manuscript&#59; critical review of the manuscript&#46;</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&#46;</p></span></span>"
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