Pleural extramedullary hematopoiesis due to severe pernicious anemia

Castillo-Torres, Sergio A.; Atilano-Díaz, Alexandro; Gómez-Almaguer, David

doi:10.1016/j.medcle.2018.09.017

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Castillo-Torres, Alexandro Atilano-Díaz, David Gómez-Almaguer" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Sergio A." "apellidos" => "Castillo-Torres" "email" => array:1 [ 0 => "sergio.castillotr@uanl.edu.mx" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Alexandro" "apellidos" => "Atilano-Díaz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "David" "apellidos" => "Gómez-Almaguer" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Universitario «Dr. José E. González», Universidad Autónoma de Nuevo León, Monterrey, Nuevo León, Mexico" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Gastroenterología, Hospital Universitario «Dr. José E. González», Universidad Autónoma de Nuevo León, Monterrey, Nuevo León, Mexico" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Hematología, Hospital Universitario «Dr. José E. González», Universidad Autónoma de Nuevo León, Monterrey, Nuevo León, Mexico" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Hematopoyesis extramedular pleural secundaria a anemia perniciosa grave" ] ] "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" => 1355 "Ancho" => 905 "Tamanyo" => 169846 ] ] "descripcion" => array:1 [ "en" => "Cytology of pleural effusion showing plasma cells (red arrow) and multisegmented neutrophils (yellow arrow) in the upper panel; erythroblasts (black arrow) and myeloblasts (white arrow) in the lower panel." ] ] ] "textoCompleto" => "Extramedullary haematopoiesis (EMH) refers to the production of viable blood cells outside the bone marrow. It occurs in a natural way during foetal development (yolk sac, liver and spleen) and pathological (infections and myeloproliferative disorders). It is diagnosed by identifying hematopoietic stem cells (cytology or biopsy); the treatment is directed to the underlying cause. The pathological form occurs most frequently in spleen and liver. It has been reported to involve virtually any organ or tissue, although the pleura is uncommon. We report the case of a 46-year-old patient with severe pernicious anemia who developed pancytopenia, pleural effusion and subacute combined degeneration, which responded to oral cobalamin supplementation.A 46-year-old male with a history of type 2 diabetes mellitus, controlled with oral antidiabetics (glycosylated haemoglobin: 5.3%). Family history of diabetes mellitus, hyperthyroidism and vitiligo. He came to be assessed due to unintentional weight loss, 6 months of progressive postural and gait instability, which required assistance to walk. On neurological examination, a pyramidal syndrome was found in the lower limbs: weakness predominantly distal (3/5), proximal (4/5), hyperreflexia (+++) and spasticity. Significant decrease in pallesthesia and proprioception, mild decrease in thermoalgesic sensitivity; broad support, high-stepping and fall gait, positive Romberg test; findings consistent with subacute combined degeneration (SCD).Pancytopenia was detected: haemoglobin 5.3g/dl, reticulocyte index 1.6%; leukocytes 3.500/mm3 and platelets 80,000/mm3; peripheral smear without evidence of multisegmented granulocytes. Lactate dehydrogenase in 7.432IU/l (upper limit: 180IU/l). Posteroanterior chest X-ray: bilateral pleural effusion. A contrast abdominal CT scan showed no malignancy data, with complete pancreatic lipomatosis, suggestive of cystic fibrosis, confirmed by a sweat chloride test (110mmol/l and 102mmol/l/48h later). Cobalamin serum levels were 115.4μmol/l (upper limit: 13.9μmol/l) and homocysteine 59pg/ml (lower limit: 211pg/ml). Bone marrow biopsy: hypercellular at the expense of the 3 hematopoietic lines, confirming the diagnosis of megaloblastic anemia. Positive serology for anti-intrinsic factor antibodies, completing diagnosis of pernicious anemia.<a class="elsevierStyleCrossRef" href="#bib0030">1</a>A thoracentesis was performed given the condition of pleural effusion without aetiology: exudate, proteins 27g/l (serum–effusion ratio: 0.48), pleural LDH 1.736IU/l (serum–effusion ratio: 0.7); cytology: 720leukocytes/mm,<a class="elsevierStyleCrossRef" href="#bib0040">3</a> with myeloblasts, erythroblasts, multisegmented neutrophils and plasma cells (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), consistent with pleural EMH. Gram stain, Ziehl-Neelsen and bacterial culture ruled out infectious causes. After partial improvement of the symptomatology, the patient was discharged with oral cobalamin supplementation: 1000μg/day.<a class="elsevierStyleCrossRef" href="#bib0035">2</a><elsevierMultimedia ident="fig0005"></elsevierMultimedia>At follow-up, one month after discharge, discrete neurological improvement (assisted walking, mild postural instability, sensory disorders); improvement of pancytopenia (haemoglobin 9.1g/dl, VCM 90.1fl; 5.700/mm leukocytes<a class="elsevierStyleCrossRef" href="#bib0040">3</a> and platelets 255,000/mm3) and lactic dehydrogenase 488UI/l (reference range: 100–200IU/l); resolution of pleural effusion and hepatosplenomegaly. A sustained haematological response was shown after one year (haemoglobin 16.3g/dl; VCM 90fl; leukocytes 8.900/mm3 and platelets 295,000/mm3) even with assisted walking.EMH produces viable cells, but never replaces haematopoiesis completely. Although normal during foetal development, in pathological cases it represents a response to processes that contribute to the bone marrow being unable to carry out normal haematopoiesis such as myelofibrosis and thalassemia. The main sites of EMH are the liver and spleen<a class="elsevierStyleCrossRef" href="#bib0040">3</a>; whereas non-hepatosplenic represents less than 5% of cases,<a class="elsevierStyleCrossRef" href="#bib0045">4</a> occurs more frequently around the spine in sites such as mediastinum, pleura, lungs, heart and pericardium. The diagnosis of pleural EMH is based on the presence of haematological stem cells in a cytology or biopsy; and treatment has focused on the underlying disease, although radiotherapy has been used in some recurrent cases or with massive effusions. For a review of pleural EMH the reader is referred to the article by Mrabti et al.<a class="elsevierStyleCrossRef" href="#bib0050">5</a>We report the first case of pleural EMH secondary to pernicious anemia, successfully treated with oral cobalamin supplementation in a patient with cystic fibrosis. We did not find case reports in which cystic fibrosis was associated with pernicious anemia. In conclusion, cobalamin deficiency should be considered in the differential diagnosis of pancytopenia, especially when other causes have been ruled out and oral supplementation of cobalamin in high doses is effective in treating pernicious anemia." "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Castillo-Torres SA, Atilano-Díaz A, Gómez-Almaguer D. Hematopoyesis extramedular pleural secundaria a anemia perniciosa grave. Med Clin (Barc). 2019;153:e7–e8." ] ] "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" => 1355 "Ancho" => 905 "Tamanyo" => 169846 ] ] "descripcion" => array:1 [ "en" => "Cytology of pleural effusion showing plasma cells (red arrow) and multisegmented neutrophils (yellow arrow) in the upper panel; erythroblasts (black arrow) and myeloblasts (white arrow) in the lower panel." ] ] ] "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" => "Clinical practice. Vitamin B12 deficiency" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "S.P. Stabler" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1056/NEJMcp1113996" "Revista" => array:6 [ "tituloSerie" => "N Engl J Med" "fecha" => "2013" "volumen" => "368" "paginaInicial" => "149" "paginaFinal" => "160" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23301732" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Oral cobalamin (vitamin B12) therapy in pernicious anemia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "E. Andres" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.autrev.2014.01.057" "Revista" => array:5 [ "tituloSerie" => "Autoimmun Rev" "fecha" => "2014" "volumen" => "13" "paginaInicial" => "778" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/24503031" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Homeostatic and pathogenic extramedullary hematopoiesis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "C.H. Kim" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.2147/JBM.S7224" "Revista" => array:6 [ "tituloSerie" => "J Blood Med" "fecha" => "2010" "volumen" => "1" "paginaInicial" => "13" "paginaFinal" => "19" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22282679" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Nonhepatosplenic extramedullary hematopoiesis: associated diseases, pathology, clinical course, and treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "C.A. Koch" 1 => "C.Y. Li" 2 => "R.A. Mesa" 3 => "A. Tefferi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.4065/78.10.1223" "Revista" => array:6 [ "tituloSerie" => "Mayo Clin Proc" "fecha" => "2003" "volumen" => "78" "paginaInicial" => "1223" "paginaFinal" => "1233" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/14531481" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pleural extramedullary hematopoiesis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "H. Mrabti" 1 => "M. Chelghoum" 2 => "L. Odier" 3 => "C. Chassagne-Clement" 4 => "M. Pavic" 5 => "Y. Devaux" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Rev Med Intern" "fecha" => "2008" "volumen" => "29" "paginaInicial" => "573" "paginaFinal" => "576" ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack459711" "titulo" => "Acknowledgements" "texto" => "The main author would like to thank Professor Matías Salinas-Chapa of the University Center for Diagnostic Imaging and Professor Olga G. Rodríguez-Cantú of the Haematology Service for their collaboration in the clinical case." "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000015300000002/v2_202004301535/S2387020619302359/v2_202004301535/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000015300000002/v2_202004301535/S2387020619302359/v2_202004301535/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020619302359?idApp=UINPBA00004N"]

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Vol. 153. Issue 2.

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Med Clin. 2024;163:e79

Medicina Clínica (English Edition) follows the Recommendations for the Conduct, Reporting, Editing and Publication of Scholarly Work in Medical Journals

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