array:23 [ "pii" => "S030028962400036X" "issn" => "03002896" "doi" => "10.1016/j.arbres.2024.01.014" "estado" => "S300" "fechaPublicacion" => "2024-05-01" "aid" => "3478" "copyright" => "SEPAR" "copyrightAnyo" => "2024" "documento" => "simple-article" "crossmark" => 1 "subdocumento" => "crp" "cita" => "Arch Bronconeumol. 2024;60:300-1" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "itemSiguiente" => array:18 [ "pii" => "S0300289624000449" "issn" => "03002896" "doi" => "10.1016/j.arbres.2024.02.005" "estado" => "S300" "fechaPublicacion" => "2024-05-01" "aid" => "3485" "copyright" => "SEPAR" "documento" => "article" "crossmark" => 1 "subdocumento" => "sco" "cita" => "Arch Bronconeumol. 2024;60:302-3" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Clinical Image</span>" "titulo" => "Thoracic Amyloidosis – A Respiratory Conundrum" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "302" "paginaFinal" => "303" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 964 "Ancho" => 1674 "Tamanyo" => 240924 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Coronal computed tomography image in soft-tissue windows showed striking peripherally calcified mediastinal and hilar lymphadenopathy. Mediastinal lymphadenopathy is usually a feature of systemic as opposed to localised disease, as demonstrated here. (B) Volume-rendered three-dimensional reconstructions highlight the extent of the lymphadenopathy shaded in blue. (C) Lung windows highlight the varied features of pulmonary parenchymal amyloidosis including septal involvement, in the form of interlobular septal thickening (<span class="elsevierStyleItalic">arrows</span>), parenchymal nodules or amyloidomas (arrowheads), peri-lymphatic calcified nodules (<span class="elsevierStyleItalic">dotted arrows</span>). (D) The patient was offered endobronchial stenting of his left main bronchus, which alleviated the symptoms greatly. (E) Mediastinal lymph node biopsy specimen obtained via endobronchial ultrasound (EBUS) demonstrates acellular deposits which appeared cyanophilic on Papanicolau's stain and eosinophilic on haematoxylin and eosin (H&E) stain. When stained with Congo red, it exhibited apple-green birefringence under polarised light characteristic of amyloidosis (the image was not available for publication).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Mohamed Tofeig, Gregory Cross, Monika Radike" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Mohamed" "apellidos" => "Tofeig" ] 1 => array:2 [ "nombre" => "Gregory" "apellidos" => "Cross" ] 2 => array:2 [ "nombre" => "Monika" "apellidos" => "Radike" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289624000449?idApp=UINPBA00003Z" "url" => "/03002896/0000006000000005/v1_202405030736/S0300289624000449/v1_202405030736/en/main.assets" ] "itemAnterior" => array:18 [ "pii" => "S0300289624000140" "issn" => "03002896" "doi" => "10.1016/j.arbres.2024.01.011" "estado" => "S300" "fechaPublicacion" => "2024-05-01" "aid" => "3475" "copyright" => "SEPAR" "documento" => "simple-article" "crossmark" => 1 "subdocumento" => "crp" "cita" => "Arch Bronconeumol. 2024;60:298-9" "abierto" => array:3 [ "ES" => false "ES2" => false "LATM" => false ] "gratuito" => false "lecturas" => array:1 [ "total" => 0 ] "en" => array:10 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>" "titulo" => "A Rare Pulmonary Metastasis of Uveal Melanoma Diagnosed Through Endobronchial Endosonography and Fine Needle Aspiration: A Case Report" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "298" "paginaFinal" => "299" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 683 "Ancho" => 1255 "Tamanyo" => 83242 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A-B: Chest CT at the time of diagnosis. C-D: Thoracic ultrasound at 4 months from diagnosis. Note the tunnelized pleural catheter in image 4 (red arrow).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Borja Domingo Cardenal, Pere Serra-Mitjà" "autores" => array:2 [ 0 => array:2 [ "nombre" => "Borja Domingo" "apellidos" => "Cardenal" ] 1 => array:2 [ "nombre" => "Pere" "apellidos" => "Serra-Mitjà" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289624000140?idApp=UINPBA00003Z" "url" => "/03002896/0000006000000005/v1_202405030736/S0300289624000140/v1_202405030736/en/main.assets" ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>" "titulo" => "Granulomatosis With Polyangiitis as a Trigger for Acute Myeloid Leukemia" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "300" "paginaFinal" => "301" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Ignacio Boira Enrique, Violeta Esteban Ronda, Eusebi Chiner Vives" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Ignacio" "apellidos" => "Boira Enrique" "email" => array:1 [ 0 => "nachoboiraenrique@hotmail.es" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Violeta" "apellidos" => "Esteban Ronda" ] 2 => array:2 [ "nombre" => "Eusebi" "apellidos" => "Chiner Vives" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Pneumology Department, University Hospital of Saint John of Alicante, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 553 "Ancho" => 1074 "Tamanyo" => 56515 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Posteroanterior and lateral chest radiograph with consolidation in the right upper lobe (white arrow) and left lower lobe (yellow arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We are presenting a case of granulomatosis with polyangiitis (GPA) triggering acute myeloid leukemia (AML).</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 38-year-old woman, without previous diseases, went to emergency department (ED) for fever, dyspnea and anosmia and she had these symptoms during the last 10 days. Chest X-ray showed consolidations in the right upper lobe and left lower lobe (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Also, laboratory tests revealed neutrophilia (8.17<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L) without leukocytosis or eosinophilia and as well as elevated acute phase reactants, without renal failure.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Thoracic computed tomography (CT) confirmed the radiographic findings and manifested a necrotic core in consolidation in the right upper lobe (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A and B). Moreover, CT of paranasal sinuses evidenced extensive sinus occupation (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>C). Microbiological cultures were negative. Antineutrophil cytoplasmic antibodies with cytoplasmic pattern (c-ANCA) were positive at 1/320 titer and anti-proteinase 3 (PR3) antibodies had a concentration of 60<span class="elsevierStyleHsp" style=""></span>IU/mL (normal is less than 10<span class="elsevierStyleHsp" style=""></span>IU/mL). In addition, a fibrobronchoscopy was performed with transbronchial biopsies showing vasculitis and granulomas with fibrinoid necrosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>D). After obtaining 13 points of the 2022 ACR/EULAR diagnostic criteria, the diagnosis of GPA was established (score ≥5). So, induction treatment with prednisone 30<span class="elsevierStyleHsp" style=""></span>mg/24<span class="elsevierStyleHsp" style=""></span>h was started and rituximab 604<span class="elsevierStyleHsp" style=""></span>mg weekly was administered weekly for 4 weeks. After three months, the patient went to ED for asthenia presenting leukocytosis (19.2<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L) with monocytosis (8.36<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L). Peripheral blood morphology evidenced 48% of blasts confirming the diagnosis of AML.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Hematologic malignancies and ANCA-associated vasculitis (AAVs) are related. Several studies have reported the increased incidence of bladder carcinoma, AML, cutaneous squamous cell carcinoma, lung cancer, colorectal carcinoma and lymphoma in AAVs due to immunosuppression, treatment and tissue inflammation. Furthermore, AAVs may present as paraneoplastic vasculitis.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Cyclophosphomide has been replaced by rituximab in induction and maintenance treatment of AAVs because of its dose-dependent oncogenicity (skin cancer, myeloid malignancies and bladder cancer).<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Rituximab increases the risk of infection, produces hypogammaglobulinemia and neutropenia, but cumulative exposure does not increase the risk of malignancy.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">3–5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Overall cancer risk is higher in patients with GPA than other AAVs. It can be explained by PR-3 which is an autoantigen at the crosstalk of autoimmunity and hematopoietic proliferation and is overexpressed in several hematological malignancies.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> PR3 is the target antigen for c-ANCA and is essential for associating GPA and hematological malignancies. It is endothelially expressed on mature neutrophils and monocytes and is inhibited by alpha-1 antitrypsin. It induces endothelial apoptosis, angiogenesis, regulation of myeloid lineage differentiation and inhibition of T-lymphocyte proliferation. In hematological malignancies, there is overexpression of PR3 due to the action of G-CSF and defects in its suppression.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The association of GPA with AML has been described in few cases in the literature, mainly in paraneoplastic vasculitis and patients treated with cyclophosphamide. In our case, the patient does not present initial alterations in the hemogram, so it is not a paraneoplastic syndrome and the AML was later, which confirms the exceptionality of the case. PR3 plays a key role in the hematological oncogenesis of GPA. The exceptional evolution presented in this case shows us the importance of close follow-up in search of hematologic malignancies in patients with AAVs.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0040" class="elsevierStylePara elsevierViewall">This work has not received funding of any kind.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of Interests</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors state that they have no conflict of interests.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of Interests" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 553 "Ancho" => 1074 "Tamanyo" => 56515 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Posteroanterior and lateral chest radiograph with consolidation in the right upper lobe (white arrow) and left lower lobe (yellow arrow).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1635 "Ancho" => 1674 "Tamanyo" => 388505 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A) Thoracic computed tomography (CT) with consolidation in the posterior segment of the right upper lobe with necrotic core (white arrow). (B) Thoracic CT with consolidation in the medial basal segment of the left lower lobe (yellow arrow). (C) CT of paranasal sinuses with complete occupation of both maxillary sinuses. (D) Histologic section of transbronchial biopsy in posterior segment of right upper lobe with presence of lung parenchyma with air spaces replaced by inflammatory cellularity of lymphohistiocytic predominance, granuloma (black arrow) and vascular inflammation with fibrinoid necrosis (white arrow).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Leukemia and myelodysplastic syndrome in granulomatosis with polyangiitis: subtypes, clinical characteristics, and outcome" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Knight" 1 => "K. Hjorton" 2 => "C. Sundström" 3 => "M. Höglund" 4 => "C. Backlin" 5 => "K.E. 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Journal Information
Case Report
Granulomatosis With Polyangiitis as a Trigger for Acute Myeloid Leukemia
Ignacio Boira Enrique
, Violeta Esteban Ronda, Eusebi Chiner Vives
Corresponding author
Pneumology Department, University Hospital of Saint John of Alicante, Spain