Letter to the Editor
Antineutrophil Cytoplasmic Antibodies (ANCA)-Negative Vasculitis in a Patient With Alpha-1-Antitrypsin Deficiency
Vasculitis con Anticuerpos anticitoplasma de neutrófilos (ANCA) negativos en paciente con déficit de alfa-1 antitripsina
Juan Marco Figueira Gonçalves
, Rosella D’amato
Corresponding author
Servicio de Neumología, Hospital Universitario Nuestra Señora de la Candelaria, Santa Cruz de Tenerife, Tenerife, Spain
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He was referred from the respiratory medicine clinic with dyspnea on moderate effort (mMRC 2). Lung function tests showed FEV<span class="elsevierStyleInf">1</span>/FVC 47%; FEV<span class="elsevierStyleInf">1</span> 1.4 l (44%); FVC 3.3 l (82%); VR 4.4 l (182%); TLC 8.0 l (119%); DLCO 49% and KCO 59%. Chest computed tomography showed severe panacinar emphysema, primarily in the lower lobes. Severe alpha-1-antitrypsin (AAT) deficiency (28.4<span class="elsevierStyleHsp" style=""></span>mg/dl) associated with ZZ phenotype was observed. A diagnosis of COPD with severe airflow obstruction and type ZZ alpha-1-antitrypsin deficiency (AATD) was made, and after abdominal ultrasound confirmed chronic liver disease, the decision was made to administer replacement AAT. Before initiation of treatment, the patient had an episode of epistaxis associated with purplish lesions in the lower limbs, tending to converge, with no blanchability on diascopy. Pathology study results showed leukocytoclastic vasculitis in small and medium caliber vessels, associated with elevated IgA (754<span class="elsevierStyleHsp" style=""></span>mg/dl), microhematuria and proteinuria suggestive of nephritis. Together, these signs were consistent with a diagnosis of adult Henoch–Schönlein purpura (HSP). ANCA antibodies (MPO<0.8<span class="elsevierStyleHsp" style=""></span>IU/ml and anti-PR3<0.4<span class="elsevierStyleHsp" style=""></span>IU/ml) and ENA were negative; ANA were positive with a titer of 1/160 in a fine speckled pattern. On the basis of this diagnosis, treatment began with oral corticosteroids (0.5<span class="elsevierStyleHsp" style=""></span>mg/kg/day) in a tapering regimen.</p><p id="par0010" class="elsevierStylePara elsevierViewall">One month after beginning this treatment, the patient suffered a fall at home and injured his left arm, with subsequent development of diffuse arthralgia and asthenia. Magnetic resonance imaging revealed cellulitis, arthritis and synovitis in the distal radioulnar and carpometacarpal joints of the upper left limb. Synovial biopsy and fluid culture were performed, confirming synovitis with positive multi-resistant <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span> culture. Despite the administration of wide-spectrum antibiotics and systemic corticosteroids, the patient developed multiorgan failure and died in the intensive care unit.</p><p id="par0015" class="elsevierStylePara elsevierViewall">A review of AATD and concomitant necrotizing vasculitis shows that microscopic polyangiitis and Wegener's granulomatosis are the most common forms, while HSP is an unusual manifestation. The mean age for presentation is generally around 48 years, and there is no prevalence with regard to sex. All cases present skin involvement, along with kidney or joint disease. In cases of HSP, hepatic cirrhosis is usually observed.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Our patient presented the foregoing conditions, further complicated by severe sepsis due to cellulitis and arthritis of the wrist. Evidence shows that AATD patients can occasionally develop necrotizing panniculitis induced by injury.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">3</span></a> In our case, the histological diagnosis could not be confirmed, since a synovial biopsy, rather than a skin biopsy, was obtained, but the clinical signs and symptoms and progress were consistent with this diagnosis.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Gonçalves JMF, D’amato R. Vasculitis con Anticuerpos anticitoplasma de neutrófilos (ANCA) negativos en paciente con déficit de alfa-1 antitripsina. Arch Bronconeumol. 2016;52:172–173.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:3 [ 0 => array:3 [ "identificador" => "bib0020" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Primary systemic vasculitis with severe α1-antitrypsin deficiency revisited" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "A. Mohammad" 1 => "M. Segelmark" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.3109/03009742.2013.846405" "Revista" => array:6 [ "tituloSerie" => "Scand J Rheumatol" "fecha" => "2014" "volumen" => "43" "paginaInicial" => "242" "paginaFinal" => "245" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/24313382" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0025" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Systemic necrotizing vasculitides in severe alpha1-antitrypsin deficiency" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "P. Mazodier" 1 => "A.N.Y. Elzouki" 2 => "M. Segelmark" 3 => "S. Eriksson" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "QJM" "fecha" => "1996" "volumen" => "89" "paginaInicial" => "599" "paginaFinal" => "611" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/8935481" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0030" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Alpha-1-antitrypsin deficiency panniculitis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "R. Valverde" 1 => "B. Rosales" 2 => "F.J. Ortiz-de Frutos" 3 => "J.L. Rodríguez-Peralto" 4 => "P.L. Ortiz-Romero" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.det.2008.05.001" "Revista" => array:6 [ "tituloSerie" => "Dermatol Clin" "fecha" => "2008" "volumen" => "26" "paginaInicial" => "447" "paginaFinal" => "451" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18793976" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/15792129/0000005200000003/v1_201602270250/S1579212916000112/v1_201602270250/en/main.assets" "Apartado" => array:4 [ "identificador" => "45358" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/15792129/0000005200000003/v1_201602270250/S1579212916000112/v1_201602270250/en/main.pdf?idApp=UINPBA00003Z&text.app=https://www.archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1579212916000112?idApp=UINPBA00003Z" ]
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