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Images: Secondary pulmonary alveolar proteinosis in brucellosis
a Department of Laboratory Medicine, Nanfang Hospital, Southern Medical University, Guangzhou 510515, China
b Department of Clinical Laboratory, Inner Mongolia Baogang Hospital, Baotou, Inner Mongolia, China
c Department of Laboratory Medicine, Hainan General Hospital/Hainan Affiliated Hospital of Hainan Medical University, Haikou, China
d Department of Laboratory Medicine, Nanjing Lishui District Hospital of Traditional Chinese Medicine, Nanjing, People's Republic of China
e Department of Clinical Laboratory, South China Hospital, Health Science Center, Shenzhen University, Shenzhen, 518116, PR China
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"apellidos" => "Yan" "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0001" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0002" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">1</span>" "identificador" => "fn1" ] ] ] 1 => array:3 [ "nombre" => "Z." "apellidos" => "Wang" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0003" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">1</span>" "identificador" => "fn1" ] ] ] 2 => array:4 [ "nombre" => "J." 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"apellidos" => "Liu" "email" => array:1 [ 0 => "liujinlinhz@163.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0001" ] ] ] ] "afiliaciones" => array:5 [ 0 => array:3 [ "entidad" => "Department of Laboratory Medicine, Nanfang Hospital, Southern Medical University, Guangzhou 510515, China" "etiqueta" => "a" "identificador" => "aff0001" ] 1 => array:3 [ "entidad" => "Department of Clinical Laboratory, Inner Mongolia Baogang Hospital, Baotou, Inner Mongolia, China" "etiqueta" => "b" "identificador" => "aff0002" ] 2 => array:3 [ "entidad" => "Department of Laboratory Medicine, Hainan General Hospital/Hainan Affiliated Hospital of Hainan Medical University, Haikou, China" "etiqueta" => "c" "identificador" => "aff0003" ] 3 => array:3 [ "entidad" => "Department of Laboratory Medicine, Nanjing Lishui District Hospital of Traditional Chinese Medicine, Nanjing, People's Republic of China" "etiqueta" => "d" "identificador" => "aff0004" ] 4 => array:3 [ "entidad" => "Department of Clinical Laboratory, South China Hospital, Health Science Center, Shenzhen University, Shenzhen, 518116, PR China" "etiqueta" => "e" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0001" "etiqueta" => "⁎" "correspondencia" => "Corresponding authors." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0001" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2002 "Ancho" => 3333 "Tamanyo" => 735066 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "alt0001" "detalle" => "Fig " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spara001" class="elsevierStyleSimplePara elsevierViewall">Secondary pulmonary alveolar proteinosis in brucellosis in a herdsman. (A) CT reveals a crazy-paving appearance on the bilateral lungs. (B) Phospholipid-rich protein aggregates are clearly observed on the BALF smear through a direct smear for microscopic examination (1000×). (C) Wright–Giemsa staining reveals bluish-purple staining (1000×). (D) Periodic acid-Schiff-positive proteinaceous material is observed (1000×). (E) Oil red O staining reveals an orange aggregate (1000×).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="para0002" class="elsevierStylePara elsevierViewall">Pulmonary alveolar proteinosis (PAP) is a rare pulmonary disease with specific features caused by the alveolar accumulation of surfactant, composed of proteins and lipids, due to dysfunctional pulmonary macrophages.<a class="elsevierStyleCrossRef" href="#bib0001"><span class="elsevierStyleSup">1</span></a> It is classified into two types primary PAP and PAP secondary to leukemia, lung infections, and inhalation of mineral particles or chemical material.<a class="elsevierStyleCrossRef" href="#bib0002"><span class="elsevierStyleSup">2</span></a> Secondary PAP (sPAP) is mainly caused by hematological disorders; sPAP associated with brucellosis is extremely rare. Here, we report a rare case of sPAP in brucellosis in a herdsman. Clinicians should consider the possibility of sPAP when chest radiography reveals abnormal findings and the bronchoalveolar lavage fluid (BALF) is milky.</p><p id="para0003" class="elsevierStylePara elsevierViewall">A 41-year-old male herdsman was hospitalized due to repeated cough and expectoration for 5 years, aggravated with shortness of breath for 5 months. Seven months prior, he was diagnosed with brucellosis with the presentation of lung infection at a local hospital, which improved after one month of treatment, leading to his discharge. His vital signs were normal. Physical examination and routine blood tests revealed unremarkable findings except for the clubbing of his digits. The electrocardiogram was negative, but a lung CT result revealed scattered patchy and large fuzzy shadows (crazy-paving appearance) in the bilateral lungs (<a class="elsevierStyleCrossRef" href="#fig0001">Fig. 1</a>A). Simultaneously, bronchoscopy was performed, and the BALF revealed a characteristic milky appearance. Interestingly, after centrifuging the BALF sample at 1500 rpm for 5 min and using the cell pellet to make a smear, a large number of phospholipid-rich protein aggregates were easily observed for microscopic examination (<a class="elsevierStyleCrossRef" href="#fig0001">Fig. 1</a>B). Moreover, the Wright–Giemsa staining for the above cell pellet smear showed bluish-purple staining (<a class="elsevierStyleCrossRef" href="#fig0001">Fig. 1</a>C). Also, periodic acid-Schiff (PAS) staining revealed PAS-positive proteinaceous material on the smear (<a class="elsevierStyleCrossRef" href="#fig0001">Fig. 1</a>D). Moreover, after incubating 100 uL oil red O with 1 mL of the BALF sample for 10 min and centrifuging the stained sample at 1500 rpm for 5 min, we used the cell pellet to make a smear, which clearly revealed an orange aggregate (<a class="elsevierStyleCrossRef" href="#fig0001">Fig. 1</a>E). Consequently, PAP was suspected. However, a test for serum anti-granulocyte macrophage colony-stimulating factor (GM-CSF) antibody yielded negative results; therefore, he was diagnosed with sPAP in brucellosis, and received several courses of whole lung lavage, his condition improved, and he was discharged.</p><elsevierMultimedia ident="fig0001"></elsevierMultimedia><p id="para0004" class="elsevierStylePara elsevierViewall">Recently, abnormalities in GM-CSF signaling are implicated in the pathogenesis of autoimmune PAP, which accounts for the vast majority of cases. However, sPAP is a rarer disorder, is not dependent on GM-CSF, and mainly occurs owing to a hematological disease.<a class="elsevierStyleCrossRef" href="#bib0003"><span class="elsevierStyleSup">3</span></a></p><p id="para0005" class="elsevierStylePara elsevierViewall">To our knowledge, this is the first report of PAP with a recent brucellosis history and GM-CSF antibody negativity. The herdsman was diagnosed with sPAP in brucellosis. Moreover, our study suggests that if the crazy-paving appearance on CT or milky BALF are observed and characteristic globules of PAS-positive proteinaceous material are also observed on the BALF, PAP should be considered as a differential diagnosis.</p><span id="sec0001" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0003">Ethical considerations</span><p id="para0006" class="elsevierStylePara elsevierViewall">Written informed consent was obtained from this patient.</p></span><span id="sec0002" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0004">Funding information</span><p id="para0007" class="elsevierStylePara elsevierViewall">None.</p></span><span id="sec0003" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0005">Consent from all authors</span><p id="para0008" class="elsevierStylePara elsevierViewall">All authors reviewed this manuscript and agreed to submit this manuscipt.</p></span><span id="sec0003a" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="cesectitle0005a">CRediT authorship contribution statement</span><p id="para0008a" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">L. Yan:</span> Writing – original draft, Visualization. <span class="elsevierStyleBold">Z. Wang:</span> Formal analysis, Writing – review & editing. <span class="elsevierStyleBold">J. Zhao:</span> Formal analysis, Writing – review & editing. <span class="elsevierStyleBold">J. Liu:</span> Writing – original draft, Formal analysis.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:5 [ 0 => array:2 [ "identificador" => "sec0001" "titulo" => "Ethical considerations" ] 1 => array:2 [ "identificador" => "sec0002" "titulo" => "Funding information" ] 2 => array:2 [ "identificador" => "sec0003" "titulo" => "Consent from all authors" ] 3 => array:2 [ "identificador" => "sec0003a" "titulo" => "CRediT authorship contribution statement" ] 4 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2022-09-06" "fechaAceptado" => "2022-09-12" "NotaPie" => array:1 [ 0 => array:3 [ "etiqueta" => "1" "nota" => "<p class="elsevierStyleNotepara" id="notep0001">These authors contributed equally to this work.</p>" "identificador" => "fn1" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0001" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2002 "Ancho" => 3333 "Tamanyo" => 735066 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "alt0001" "detalle" => "Fig " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spara001" class="elsevierStyleSimplePara elsevierViewall">Secondary pulmonary alveolar proteinosis in brucellosis in a herdsman. (A) CT reveals a crazy-paving appearance on the bilateral lungs. (B) Phospholipid-rich protein aggregates are clearly observed on the BALF smear through a direct smear for microscopic examination (1000×). (C) Wright–Giemsa staining reveals bluish-purple staining (1000×). (D) Periodic acid-Schiff-positive proteinaceous material is observed (1000×). (E) Oil red O staining reveals an orange aggregate (1000×).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "cebibsec1" "bibliografiaReferencia" => array:3 [ 0 => array:3 [ "identificador" => "bib0001" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Crazy paving in pulmonary alveolar proteinosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "B.W. Allwood" 1 => "S. Bennji" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1056/NEJMicm1908563" "Revista" => array:6 [ "tituloSerie" => "N Engl J Med" "fecha" => "2020" "volumen" => "382" "numero" => "3" "paginaInicial" => "275" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31940701" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0002" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary alveolar proteinosis: crazing-paving appearance" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "H. Matsuura" 1 => "Y. Yamaji" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.amjmed.2017.11.013" "Revista" => array:7 [ "tituloSerie" => "Am J Med" "fecha" => "2018" "volumen" => "131" "numero" => "4" "paginaInicial" => "e153" "paginaFinal" => "e154" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/29191486" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0003" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Secondary pulmonary alveolar proteinosis in acute myeloid leukemia" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:3 [ 0 => "T. Hirakawa" 1 => "M. Taniwaki" 2 => "M. Yamasaki" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "QJM" "fecha" => "2019" "volumen" => "112" "numero" => "4" "paginaInicial" => "293" "paginaFinal" => "294" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/25310437/0000002900000004/v1_202307031326/S2531043722002215/v1_202307031326/en/main.assets" "Apartado" => array:4 [ "identificador" => "88777" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Images" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/25310437/0000002900000004/v1_202307031326/S2531043722002215/v1_202307031326/en/main.pdf?idApp=UINPBA00004E&text.app=https://journalpulmonology.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2531043722002215?idApp=UINPBA00004E" ]
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