Case Report
Lung Cancer With Cold Agglutinin Syndrome as a Paraneoplastic Syndrome: A Case Report
Pedro Pereira Barros
, Bruno dos Santos, Ulisses Brito
Corresponding author
Centro Hospitalar Universitário do Algarve – Hospital de Faro, Portugal
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Bronchoscopy showed chronic inflammation (B). Paranasal sinus CT image with negative result (C). Chest CT reexamination images showed the consolidation in the right middle lobe (D). Bronchoscopy showed a yellow mucous plugs, in the right middle lobe (E) and complete removed by cryotherapy under bronchoscopy (F). Eosinophils count and total serum IgE level (G). Chest CT 2 weeks after treatment with VRCZ (H). Chest CT 6 months after treatment with VRCZ (I).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Xiaoting Zhou, Jiangnan Zheng, Jianfeng Zhang" "autores" => array:3 [ 0 => array:2 [ "nombre" => "Xiaoting" "apellidos" => "Zhou" ] 1 => array:2 [ "nombre" => "Jiangnan" "apellidos" => "Zheng" ] 2 => array:2 [ "nombre" => "Jianfeng" "apellidos" => "Zhang" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289622005774?idApp=UINPBA00003Z" "url" => "/03002896/0000005900000002/v3_202307071534/S0300289622005774/v3_202307071534/en/main.assets" ] "itemAnterior" => array:18 [ "pii" => "S0300289622005257" "issn" => "03002896" "doi" => "10.1016/j.arbres.2022.09.004" "estado" => "S300" "fechaPublicacion" => "2023-02-01" "aid" => "3174" "copyright" => "SEPAR" "documento" => "article" "crossmark" => 1 "subdocumento" => "ssu" "cita" => "Arch Bronconeumol. 2023;59:101-8" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "en" => array:12 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Special Article</span>" "titulo" => "Pathophysiology of Chronic Bronchial Infection in Bronchiectasis" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "101" "paginaFinal" => "108" ] ] "contieneResumen" => array:1 [ "en" => true ] "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" => 1671 "Ancho" => 2508 "Tamanyo" => 362126 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Interaction between microbiological agents and airway epithelium in bronchiectasis. The bronchial epithelium in bronchiectasis is disrupted and shows increased mucus hypersecretion, especially in the presence of bronchial infection. Bacteria such as <span class="elsevierStyleItalic">Haemophilus influenzae</span>, <span class="elsevierStyleItalic">Streptococcus pneumoniae</span>, <span class="elsevierStyleItalic">Streptococcus aureus</span> and <span class="elsevierStyleItalic">Pseudomonas aeruginosa</span> are often the causative agents of bronchial infections. Fungi such as <span class="elsevierStyleItalic">Candida albicans</span> and <span class="elsevierStyleItalic">Aspergillus fumigatus</span> are other microbes that can cause bronchial infection. Non-tuberculous mycobacteria (NTM) can directly cause bronchiectasis or contribute to worsening previous pathology. The airway epithelium recruits neutrophils, macrophages and eosinophils that migrate into the airways and participate in host defence. <span class="elsevierStyleItalic">P. aeruginosa</span> often forms biofilms to evade the immune system. Another mechanism of immune evasion is bypassing the neutrophil extracellular traps (NETs).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Belén Solarat, Lidia Perea, Rosa Faner, David de La Rosa, Miguel Ángel Martínez-García, Oriol Sibila" "autores" => array:6 [ 0 => array:2 [ "nombre" => "Belén" "apellidos" => "Solarat" ] 1 => array:2 [ "nombre" => "Lidia" "apellidos" => "Perea" ] 2 => array:2 [ "nombre" => "Rosa" "apellidos" => "Faner" ] 3 => array:2 [ "nombre" => "David" "apellidos" => "de La Rosa" ] 4 => array:2 [ "nombre" => "Miguel Ángel" "apellidos" => "Martínez-García" ] 5 => array:2 [ "nombre" => "Oriol" "apellidos" => "Sibila" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289622005257?idApp=UINPBA00003Z" "url" => "/03002896/0000005900000002/v3_202307071534/S0300289622005257/v3_202307071534/en/main.assets" ] "en" => array:13 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>" "titulo" => "Lung Cancer With Cold Agglutinin Syndrome as a Paraneoplastic Syndrome: A Case Report" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "109" "paginaFinal" => "110" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Pedro Pereira Barros, Bruno dos Santos, Ulisses Brito" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Pedro" "apellidos" => "Pereira Barros" "email" => array:1 [ 0 => "pedro34621@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Bruno" "apellidos" => "dos Santos" ] 2 => array:2 [ "nombre" => "Ulisses" "apellidos" => "Brito" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Centro Hospitalar Universitário do Algarve – Hospital de Faro, Portugal" "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" => 2007 "Ancho" => 2007 "Tamanyo" => 291932 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest X-ray showing a mass in the right upper lobe; (B) hepatic nodule identified in the abdominal CT scan; (C) cranioencephalic CT scan with the presence of a nodule in the occipital area, with vasogenic oedema; (D) abdominal CT scan showing a thrombosed aneurysm in the abdominal aorta.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">62-Year-old male, smoker (100 Pack-year), presented with dry cough and dizziness within the past 3 week, being hospitalized after detecting a lung mass in the right upper lobe in the chest X-ray (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). CT scan confirmed the presence of the lung mass, also identifying bilateral lung nodules, mediastinal adenopathies, and a hepatic nodule (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). Cranioencephalic CT scan found multiple brain nodules, associated with vasogenic oedema (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). A transthoracic needle biopsy was performed, identifying a lung adenocarcinoma with negative molecular study and PD-L1 of 1–5%.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was discharged and 3 weeks later he presented with asthenia, haematuria, constipation, and jaundice. Laboratory tests revealed severe macrocytic anaemia with 4.2<span class="elsevierStyleHsp" style=""></span>g/dL haemoglobin (13.5<span class="elsevierStyleHsp" style=""></span>g/dL haemoglobin at discharge), and leucocytosis in the complete blood count. Elevated levels of LDH (1069<span class="elsevierStyleHsp" style=""></span>UI/L), bilirubin (6<span class="elsevierStyleHsp" style=""></span>mg/dL), and C-reactive protein (362<span class="elsevierStyleHsp" style=""></span>mg/dL) were also found. After evaluation by immunohemotherapy, irregular antibody screening was positive, with a positive direct Coombs test and C3d monospecific test. These results sustained the diagnosis of an autoimmune haemolytic anaemia due to cold agglutinin syndrome. The patient was started on antibiotics, in addition to corticotherapy, IV immunoglobulin on 2 consecutive days, and transfusion of 4 units of heated red blood cell concentrates, while maintaining body temperature above 35<span class="elsevierStyleHsp" style=""></span>°C. Blood and urine cultures identified Methicillin-Sensitive Staphylococcus aureus (MSSA).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Two weeks after being discharged, the patient presented asthenia, anorexia, and joint pain. Blood cultures were again positive for MSSA, and an abdominal CT identified a thrombosed abdominal aortic aneurysm (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D), where a possible lodging for the bacteria was suspected. After two cycles of antibiotics, blood cultures were negative and haemoglobin values were stable after transfusing one unit of heated red blood cell concentrate. Chemotherapy with Carboplatin and Pemetrexed was started during hospitalization. After this episode, the patient experienced sudden pain and swelling in his left leg. Echo-doppler identified thrombi in the deep veins bilaterally, causing subtotal obliteration on the right side and complete on the left. Analytically, he presented pancytopenia, requiring a new transfusion under steroid therapy and IV immunoglobulin. Chemotherapy was later changed to Vinorelbine, administering the first cycle during hospitalization. There was a progressive deterioration in the patient's general condition, culminating in anuria and prostration one month after the last hospitalization, ultimately leading up to the patient's death.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Paraneoplastic syndromes are rare disorders in patients with solid tumours, triggered by remote effects of the tumour, such as an ectopic production of biological substances or by abnormal immune system response to the tumour.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> Autoimmune hemolytic anaemia is a known paraneoplastic phenomenon.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Cold agglutinin syndrome is a rare type of autoimmune hemolytic anaemia, produced by antibodies that react at low temperatures with the patient's own erythrocytes, destroying them. This syndrome is mostly associated with lymphoproliferative disorders, although there have been reported cases of paraneoplastic cold agglutinin syndrome associated with solid tumours, including lung adenocarcinoma.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">In this case, the appearance of this syndrome triggered frequent complications, leading to a deterioration of the patient's general condition and several hospitalizations, which made the approach and treatment of the neoplasia very challenging.</p><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0035" class="elsevierStylePara elsevierViewall">There is no funding related to this article.</p></span><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interests</span><p id="par0030" 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" => "sec0010" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2007 "Ancho" => 2007 "Tamanyo" => 291932 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Chest X-ray showing a mass in the right upper lobe; (B) hepatic nodule identified in the abdominal CT scan; (C) cranioencephalic CT scan with the presence of a nodule in the occipital area, with vasogenic oedema; (D) abdominal CT scan showing a thrombosed aneurysm in the abdominal aorta.</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" => "Paraneoplastic humorally mediated hypercalcemia induced by parathyroid hormonerelated protein in gynecologic malignancies: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "P. Savvari" 1 => "P. Peitsidis" 2 => "M. Alevizaki" 3 => "M.A. Dimopoulos" 4 => "A. Antsaklis" 5 => "C.A. Papadimitriou" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1159/000226209" "Revista" => array:6 [ "tituloSerie" => "Onkologie" "fecha" => "2009" "volumen" => "32" "paginaInicial" => "517" "paginaFinal" => "523" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/19745599" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Paraneoplastic disorders" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "E. Lancaster" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1212/CON.0000000000000542" "Revista" => array:5 [ "tituloSerie" => "Continuum (Minneap Minn) [Internet]" "fecha" => "2017" "volumen" => "23" "paginaInicial" => "1653" "paginaFinal" => "1679" ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Autoimmune hemolytic anemia as a paraneoplastic phenomenon in solid tumors: a critical analysis of 52 cases reported in the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J. Puthenparambil" 1 => "K. Lechner" 2 => "G. Kornek" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s00508-010-1319-z" "Revista" => array:6 [ "tituloSerie" => "Wien Klin Wochenschr" "fecha" => "2010" "volumen" => "122" "paginaInicial" => "229" "paginaFinal" => "236" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/20503022" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cold agglutinin autoimmune hemolytic anemia in nonhematologic malignancies" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J. Wortman" 1 => "W. Rosse" 2 => "G. Logue" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1002/ajh.2830060312" "Revista" => array:6 [ "tituloSerie" => "Am J Hematol" "fecha" => "1979" "volumen" => "6" "paginaInicial" => "275" "paginaFinal" => "283" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/484548" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A novel approach for treatment of cold agglutinin syndrome-related severe hemolysis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "A. Tesfaye" 1 => "C. Broome" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.14740/jh242w" "Revista" => array:5 [ "tituloSerie" => "J Hematol (Brossard)" "fecha" => "2016" "volumen" => "5" "paginaInicial" => "30" "paginaFinal" => "33" ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/03002896/0000005900000002/v3_202307071534/S0300289622005919/v3_202307071534/en/main.assets" "Apartado" => array:4 [ "identificador" => "95260" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Case Report" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/03002896/0000005900000002/v3_202307071534/S0300289622005919/v3_202307071534/en/main.pdf?idApp=UINPBA00003Z&text.app=https://archbronconeumol.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0300289622005919?idApp=UINPBA00003Z" ]
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