What is your diagnosis?
Case for diagnosis. Cutaneous small vessel vasculitis (anti-proteinase 3 positive), fever, hemoptysis, and lung cavitation in an adult
Luana Moraes Campos, Mariana Righetto de Ré, Priscila Neri Lacerda, Hélio Amante Miot
Corresponding author
Department of Dermatology and Radiotherapy, Faculty of Medicine, Universidade Estadual Paulista, Botucatu, SP, Brazil
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(A) S-100 was negative in the tumor cells, while the nerve fiber tract served as an internal positive control (Original magnification, ×200). 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Cutaneous small vessel vasculitis (anti-proteinase 3 positive), fever, hemoptysis, and lung cavitation in an adult" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "240" "paginaFinal" => "242" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Luana Moraes Campos, Mariana Righetto de Ré, Priscila Neri Lacerda, Hélio Amante Miot" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Luana Moraes" "apellidos" => "Campos" ] 1 => array:2 [ "nombre" => "Mariana Righetto" "apellidos" => "de Ré" ] 2 => array:2 [ "nombre" => "Priscila Neri" "apellidos" => "Lacerda" ] 3 => array:4 [ "nombre" => "Hélio Amante" "apellidos" => "Miot" "email" => array:1 [ 0 => "heliomiot@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology and Radiotherapy, Faculty of Medicine, Universidade Estadual Paulista, Botucatu, SP, 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" => 563 "Ancho" => 750 "Tamanyo" => 165912 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Histopathological examination of the skin showing intense diffuse inflammatory infiltrate in the dermis, red blood cells leakage, vascular walls infiltrated by neutrophils, and fibrinoid necrosis. Subepidermal vesicle, filled with red blood cells and neutrophils (Hematoxylin & eosin, ×10).</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">47-year-old male, smoker, reported the appearance of petechiae, palpable purpura, and hemorrhagic blisters, on the soles and palms (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) a week before, progressing to the lower and upper abdomen, and face (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>); he also reported bleeding in the eyes, nose, hemoptysis, and fever. Chest tomography revealed cavitation, with thick walls in the right upper lobe, and with sparse consolidations in other pulmonary areas (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">The laboratory tests demonstrated high levels of classic anti-neutrophil cytoplasmic antibody (c-ANCA: anti-proteinase 3 > 90 U/mL [reference values: < 5 U/mL]). Other viral serologies were negative, and the markers of inflammatory tests were very high: ESR 64 mm and CRP 19 mg/L.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histopathological examination of the skin revealed leukocytoclastic vasculitis, and direct immunofluorescence was negative with anti-IgM, anti-IgG, and anti-IgA antibodies. (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>). Pulmonary histopathology showed a granulomatous inflammatory process with central caseation. The bronchoalveolar lavage revealed three alcohol-acid fast bacilli (AAFB).</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">What is your diagnosis?</span><p id="par0020" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">a)</span><p id="par0025" class="elsevierStylePara elsevierViewall">Small vessel vasculitis secondary to tuberculosis</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">b)</span><p id="par0030" class="elsevierStylePara elsevierViewall">Granulomatosis with polyangiitis (Wegener's granulomatosis [WG])</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">c)</span><p id="par0035" class="elsevierStylePara elsevierViewall">Atypical mycobacteriosis with embolization</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">d)</span><p id="par0040" class="elsevierStylePara elsevierViewall">Association of tuberculosis with Wegener's granulomatosis</p></li></ul></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0045" class="elsevierStylePara elsevierViewall">The most common manifestation of <span class="elsevierStyleItalic">Mycobacterium tuberculosis</span> infection is pulmonary; however, more than 10% of cases have an extrapulmonary presentation.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> As the incidence of tuberculosis (TB) has been increasing worldwide, this affects the occurrence of atypical forms.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Skin lesions associated with TB are highly polymorphic, and can occur by direct action of bacilli, by inoculation, hematogenous propagation, deposition of immune complexes on the walls of small vessels (hypersensitivity vasculitis), or by the formation of antibodies against antigens in the host.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–5</span></a> Small vessel vasculitis secondary to TB is uncommon, with less than 20 cases reported in the literature. Three forms have been described: Henoch-Schönlein purpura, vasculitis secondary to rifampicin, and cutaneous leukocytoclastic vasculitis.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">TB can occur associated with other autoimmune diseases, such as WG, sharing similar clinical findings and histopathology.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,7</span></a> Necrotic lung lesions in WG are radiologically similar to those observed in TB. It is worth mentioning that the literature presents two case reports in which these two diseases coexisted.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Although ANCAs are considered to be markers of systemic vasculitis and are associated with WG and other autoimmune disorders, ANCA positivity has been demonstrated in infectious diseases such as TB, especially the c-ANCA pattern, increasing the possibility of diagnostic confusion with WG.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,7,9,10</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">M. tuberculosis</span> can stimulate the release of oxygen metabolites from activated neutrophils, which would release lysosomal enzymes in the early stages of infection, with the potential to induce autoantibodies against these components.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In this case, the patient met diagnostic criteria for WG, and had high levels of c-ANCA, generating diagnostic doubt about the possibility of concomitant diseases. The absence of previous sinusopathy, asymmetry of pulmonary involvement, and lack of involvement of medium size vessels (livedo, ulcers, or necrosis of extremities), indicated an atypical form of TB.</p><p id="par0075" class="elsevierStylePara elsevierViewall">The patient was submitted to an antituberculous regimen (RIPE) and corticosteroid therapy 1 mg/kg/day, with remission of the condition after one month of treatment, and normalization of c-ANCA after six months.</p><p id="par0080" class="elsevierStylePara elsevierViewall">In countries with a high prevalence of TB, the presence of autoantibodies in a patient with vasculitis, fever, and pulmonary cavitation requires the investigation of infectious causes, especially tuberculosis, before admitting the diagnosis of WG.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,7</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0085" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors’ contribution</span><p id="par0090" class="elsevierStylePara elsevierViewall">Luana Moraes Campos: Approval of the manuscript; drafting of the manuscript; effective participation in research orientation; effective participation in propaedeutics; literature review; critical review of the manuscript.</p><p id="par0095" class="elsevierStylePara elsevierViewall">Mariana Righetto de Ré: Approval of the manuscript; drafting of the manuscript; effective participation in research orientation; effective participation in propaedeutics; literature review; critical review of the manuscript.</p><p id="par0100" class="elsevierStylePara elsevierViewall">Priscila Neri Lacerda: Approval of the manuscript; drafting of the manuscript; effective participation in research orientation; effective participation in propaedeutics; literature review; critical review of the manuscript.</p><p id="par0105" class="elsevierStylePara elsevierViewall">Hélio Amante Miot: Approval of the manuscript; drafting of the manuscript; effective participation in research orientation; effective participation in propaedeutics; literature review; critical review of the manuscript.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0110" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1505264" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1366020" "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’ contribution" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflicts of Interest" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-06-02" "fechaAceptado" => "2020-06-24" "PalabrasClave" => array:1 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1366020" "palabras" => array:4 [ 0 => "Anti-neutrophil cytoplasmic antibodies" 1 => "Anti-neutrophil cytoplasmic antibody-associated vasculitis" 2 => "Tuberculosis" 3 => "Vasculitis" ] ] ] ] "tieneResumen" => true "resumen" => array:1 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Small vessel vasculitis with anti-proteinase antibodies 3 is an atypical clinical presentation of tuberculosis. The authors present the case of a 47-year-old male patient, with palpable purpura and palmoplantar hemorrhagic blisters, with subsequent dissemination. He presented severe pulmonary symptoms with cavitation, fever, hemoptysis, and high levels of anti-proteinase 3. Histopathological assessment of the skin revealed small vessel vasculitis; pulmonary histopathology showed granulomas with caseation. Bronchoalveolar lavage was positive for alcohol-acid-fast bacilli. In countries with a high prevalence of tuberculosis, the presence of autoantibodies in a patient with vasculitis, fever, and pulmonary cavitation requires investigation of infectious causes.</p></span>" ] ] "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Campos LM, de Ré MR, Lacerda PN, Miot HA. Case for diagnosis. Cutaneous small vessel vasculitis (anti-proteinase 3 positive), fever, hemoptysis, and lung cavitation in an adult. An Bras Dermatol. 2021;96:240–2.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Department of Dermatology and Radiotherapy, School of Medicine, Universidade Estadual Paulista, Botucatu, SP, 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" => 480 "Ancho" => 750 "Tamanyo" => 49773 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple, bilateral palpable petechiae and purpura on the soles.</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" => 453 "Ancho" => 750 "Tamanyo" => 42501 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Palpable purpura and necrotic vesicles on the left lower limb.</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" => 422 "Ancho" => 750 "Tamanyo" => 47405 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Computed tomography scan of the chest showing cavitation with a thick wall in the right upper lobe amid consolidations in other pulmonary areas.</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" => 563 "Ancho" => 750 "Tamanyo" => 165912 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Histopathological examination of the skin showing intense diffuse inflammatory infiltrate in the dermis, red blood cells leakage, vascular walls infiltrated by neutrophils, and fibrinoid necrosis. Subepidermal vesicle, filled with red blood cells and neutrophils (Hematoxylin & eosin, ×10).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Tuberculosis mimicking primary systemic vasculitis: not to be missed!" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "A. Jain" 1 => "D.P. Misra" 2 => "A. Ramesh" 3 => "D. Basu" 4 => "V.K. Jain" 5 => "V.S. 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