Pancreatic lymphoma: Case report

Rubín de Célix, Cristina; Gómez-Labrador, Celia; Mendoza, Jorge

doi:10.1016/j.medcle.2020.07.032

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CRP: C-reactive protein. aInterpret with caution considering the high prevalence of asymptomatic bacteriuria in this patient population. bConsider: thyroid-stimulating hormone, procalcitonin, and blood cultures." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Pere Tudela, Sandra Clotet, Antònia Segura, Anna Carreres" "autores" => array:4 [ 0 => array:2 [ "nombre" => "Pere" "apellidos" => "Tudela" ] 1 => array:2 [ "nombre" => "Sandra" "apellidos" => "Clotet" ] 2 => array:2 [ "nombre" => "Antònia" "apellidos" => "Segura" ] 3 => array:2 [ "nombre" => "Anna" "apellidos" => "Carreres" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S238702062100468X?idApp=UINPBA00004N" "url" => "/23870206/0000015700000006/v2_202201010922/S238702062100468X/v2_202201010922/en/main.assets" ] "en" => array:16 [ "idiomaDefecto" => true "cabecera" => "Scientific letter" "titulo" => "Pancreatic lymphoma: Case report" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "298" "paginaFinal" => "299" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Cristina Rubín de Célix, Celia Gómez-Labrador, Jorge Mendoza" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Cristina" "apellidos" => "Rubín de Célix" "email" => array:1 [ 0 => "cristina.rubindecelix@salud.madrid.org" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Celia" "apellidos" => "Gómez-Labrador" ] 2 => array:2 [ "nombre" => "Jorge" "apellidos" => "Mendoza" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Gastroenterology Department, La Princesa University Hospital, Instituto de Investigación Sanitaria Princesa (IIS-IP), Centro de Investigación Biomédica en Red de Enfermedades Hepáticas y Digestivas (CIBEREHD), Madrid, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Linfoma pancreático: presentación de un caso" ] ] "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" => 1246 "Ancho" => 3167 "Tamanyo" => 456398 ] ] "descripcion" => array:1 [ "en" => "(A) EUS: rounded mass of 25mm in pancreatic head. (B) Abdominal CT: diffuse infiltration of the head and neck of the pancreas (arrow), with an edematous and hypovascular appearance. (C) PET-CT: hypermetabolic pancreatic head mass. Rupture of the cortical of the left clavicle. (D) Shoulder RM (sagittal): T1 hypointense left clavicle bone marrow, with cortical rupture. (E) BAG pancreatic lesion: Monomorphic proliferation of lymphoid cells of intermediate size. Frequent mitosis. Macrophages with cellular debris (arrows: “starry-sky” pattern). Hematoxylin–Eosin, 20×. (F) FNA of pancreatic lesion and supraclavicular lesion (G) with abundant lymphoid cellularity with little cytoplasm with small intracytoplasmic vacuoles. Diff Quick, 60×." ] ] ] "textoCompleto" => "Pancreatic involvement as a result of a lymphoma is rare in our environment (less than 2% of extranodal lymphomas). The finding of a pancreatic mass implies a wide differential diagnosis, among which is pancreatic adenocarcinoma (PCA) with an incidence of 15/100,000 inhabitants.<a class="elsevierStyleCrossRef" href="#bib0030">1</a> The case of an immunocompetent male with an onset of acute pancreatitis, pancreatic mass and clavicular bone involvement is presented.A 26-year-old man with no medical history was assessed in the Emergency Department due to a two-week history of upper abdominal discomfort and weight loss of 4kg and he reported pain of the left sternoclavicular joint that he had related to overexertion. Clinical examination revealed a non-painful abdomen and his left sternoclavicular joint was mildly painful. Laboratory tests showed a bilirubin of 6.22mg/dL, AST 385U/L, ALT 977U/L, GGT 460U/L, ALP 315U/L, LDH 347U/L and lipase 993UI. An abdominal ultrasound showed biliary sludge, a dilated bile duct and a Wirsung duct dilatation (4.7mm). Admission was decided with the initial suspicion of acute pancreatitis and obstructive jaundice. An endoscopic ultrasound (EUS) revealed an enlarged hypoechoic pancreatic parenchyma with a beaded Wirsung duct. In the pancreatic head, a rounded mass of 25mm isoechoic was identified with a greenish-blue elastographic pattern (strain ratio of 21.6). Samples were taken with a 22G histology needle for study including IgG4 to rule out a possible pseudotumor within an autoimmune pancreatitis (AIP) (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>A). Hepatotropic virus, HIV and autoimmunity serologies were negative and tumor markers were not elevated. Plasma IgG4 levels were 171mg/dL. A thoracoabdominal CT scan suggested findings of AIP, without ruling out neoplastic lymphoproliferative process (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>B). The initial pathology study showed an inflammatory infiltrate without dysplasia, and given the possibility of an autoimmune etiology, it was decided to start prednisone 40mg. After 10 days of treatment the patient presented increasing hyperbilirubinemia and it was decided perform a magnetic resonance of the shoulder that identified an aggressive bone lesion of the left clavicle, suggesting lymphoma (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>D). The immunohistochemical study revealed participation of CD20+ B lymphocytes, without IgG4 expression. A biopsy of both the clavicle and pancreas percutaneously was done and confirmed the diagnosis of Burkitt-type high-grade non-Hodgkin lymphoma (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>E–G). PET-CT showed bone infiltration with metabolic criteria for malignancy (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>C). Biliary drainage was performed by ERCP with the placement of a metallic covered prosthesis achieving a decrease in bilirubin, allowing to start treatment using the R-Hyper CVAD regimen (rituximab, cyclophosphamide, vincristine, adriamycin, dexamethasone), with an initial complete response to treatment.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>The finding of a pancreatic mass is a diagnostic challenge due to the difficulty in making the differential diagnosis between PCA, AIP o pancreatic lymphoma (PL). AIP is an inflammatory and fibrosing disease with an incidence of 1:100,000. AIP diagnosis is based on the HISORt criteria: histology (more than 10 IgG4-positive cells), imaging test, serology with IgG4 elevation ≥2 times the upper limit of normality, extrapancreatic manifestations and response to steroid treatment.<a class="elsevierStyleCrossRefs" href="#bib0035">2,3</a> In our case, the initial imaging tests made us think of AIP, however the histology was not compatible and there was no response to steroids.Gastrointestinal tract lymphomas are not uncommon, especially at the gastric and small intestine and most of them are line B non-Hodgkin lymphomas. Burkitt's Lymphoma (BL) is an aggressive lymphoma characterized by the c-myc gene dysregulation and presents itself in three variants: endemic, immunodeficiency-related and sporadic. The latter it is more frequently diagnosed in young patients with an abdominal mass. The EUS-guided puncture is the first choice technique and histology shows atypical lymphoid cells, with a “starry-sky” appearance with positive markers for CD19 and CD20. In our case, the sample obtained by EUS was artifacted, so finally the percutaneous biopsy with a needle of 18G was key in the diagnosis, demonstrating the markers and typical cellularity of BL.<a class="elsevierStyleCrossRefs" href="#bib0045">4,5</a>This case emphasizes the need to obtain a representative sample for the differential diagnosis of a pancreatic mass. Radiological and laboratory findings alone are not enough for diagnosis, and histology is essential for targeted therapy. To our knowledge, this is the first case published of BL in an immunocompetent patient who debuted in the form of bone lesion and acute pancreatitis.FundingThe authors received no specific funding for this work.Conflict of interestAuthors declare not to have any conflict of interests that affects their impartiality." 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(D) Shoulder RM (sagittal): T1 hypointense left clavicle bone marrow, with cortical rupture. (E) BAG pancreatic lesion: Monomorphic proliferation of lymphoid cells of intermediate size. Frequent mitosis. Macrophages with cellular debris (arrows: “starry-sky” pattern). Hematoxylin–Eosin, 20×. (F) FNA of pancreatic lesion and supraclavicular lesion (G) with abundant lymphoid cellularity with little cytoplasm with small intracytoplasmic vacuoles. Diff Quick, 60×." ] ] ] "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" => "Annual report to the nation on the status of cancer, Part I: National cancer statistics" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "S.J. Henley" 1 => "E.M. Ward" 2 => "S. Scott" 3 => "J. Ma" 4 => "R.N. Anderson" 5 => "A.U. Firth" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1002/cncr.32802" "Revista" => array:2 [ "tituloSerie" => "Cancer" "fecha" => "2020" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Management of autoimmune pancreatitis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "V.S. Akshintala" 1 => "V.K. Singh" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.cgh.2019.04.052" "Revista" => array:6 [ "tituloSerie" => "Clin Gastroenterol Hepatol" "fecha" => "2019" "volumen" => "17" "paginaInicial" => "1937" "paginaFinal" => "1939" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31042584" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0040" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "International consensus for the treatment of autoimmune pancreatitis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "K. Okazaki" 1 => "S.T. Chari" 2 => "L. Frulloni" 3 => "M.M. Lerch" 4 => "T. Kamisawa" 5 => "S. Kawa" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.pan.2016.12.003" "Revista" => array:6 [ "tituloSerie" => "Pancreatology" "fecha" => "2017" "volumen" => "17" "paginaInicial" => "1" "paginaFinal" => "6" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/28027896" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Burkitt and Burkitt-like lymphomas: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "K. Saleh" 1 => "J.M. Michot" 2 => "V. Camara-Clayette" 3 => "Y. Vassetsky" 4 => "V. Ribrag" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s11912-020-0898-8" "Revista" => array:5 [ "tituloSerie" => "Curr Oncol Rep" "fecha" => "2020" "volumen" => "22" "paginaInicial" => "33" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32144513" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "B-cell lymphoma presenting as acute pancreatitis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "E. Federico" 1 => "M. Falconi" 2 => "G. Zuodar" 3 => "G. Falconieri" 4 => "F. Puglisi" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1159/000332038" "Revista" => array:7 [ "tituloSerie" => "Pancreatology" "fecha" => "2011" "volumen" => "11" "paginaInicial" => "553" "paginaFinal" => "556" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/22205036" "web" => "Medline" ] ] "itemHostRev" => array:3 [ "pii" => "S0735109704018601" "estado" => "S300" "issn" => "07351097" ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack578995" "titulo" => "Acknowledgments" "texto" => "To Mariel Fabiola Valdivia, Raquel Herranz, Tomás Álvarez-Malé and Cecilio Santander who reviewed the present document." "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000015700000006/v2_202201010922/S2387020621004514/v2_202201010922/en/main.assets" "Apartado" => array:4 [ "identificador" => "43311" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Scientific letters" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000015700000006/v2_202201010922/S2387020621004514/v2_202201010922/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020621004514?idApp=UINPBA00004N"]

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Vol. 157. Issue 6.

Pages 298-299 (September 2021)

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