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Scientific letters
Intestinal Burkitt lymphoma in a patient with coeliac disease
Linfoma de Burkitt intestinal en un paciente con enfermedad celíaca
Emanuel José Saada, Carina Elizabeth Seculini Patiñoa, María Victoria Laborieb, Luciana Guanchialec, Domingo Balderramod,
Corresponding author
a Internal Medicine Department, Hospital Privado Universitario de Córdoba, Instituto Universitario de Ciencias Biomédicas de Córdoba, Argentina
b Pathology Department, Hospital Privado Universitario de Córdoba, Instituto Universitario de Ciencias Biomédicas de Córdoba, Argentina
c Hematology and Oncology Department, Hospital Privado Universitario de Córdoba, Instituto Universitario de Ciencias Biomédicas de Córdoba, Argentina
d Gastroenterology Department, Hospital Privado Universitario de Córdoba, Instituto Universitario de Ciencias Biomédicas de Córdoba, Argentina
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Intestinal Burkitt lymphoma&#46; &#40;a&#41; Axial Computed Tomography cut&#44; with a regular parietal thickening of the terminal ileum that protrudes the cecal base through the valve&#46; &#40;b&#41; Colonoscopy exam showed a 4<span class="elsevierStyleHsp" style=""></span>cm pseudopolyploid ileal lesion that protruded through ileocecal valve&#46; &#40;c&#41; H &#38; E original magnification 400&#215;&#46; A diffuse proliferation of neoplastic cells is observed from medium to large size&#44; among which macrophages can be found&#44; resulting in &#8220;starry-sky&#8221; pattern characteristic of Burkitt lymphoma&#46; &#40;d&#41; Positive staining for CD 20 in immunohistochemistry&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Non-Hodgkin lymphomas &#40;NHL&#41; represent a small percentage of all gastrointestinal tumors&#46; The major risk factors for gastrointestinal NHL include immunosuppression and celiac disease &#40;CD&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> Burkitt lymphoma &#40;BL&#41; is an infrequent aggressive variant of NHL that frequently affect the gastrointestinal tract&#46; BL is common in young children in Africa who also have malaria and Epstein&#8211;Barr virus infections&#46; The latter infection is associated with the majority of African cases&#46; Outside of Africa&#44; BL is infrequent and is especially related to human immunodeficiency virus &#40;HIV&#41; infection&#46; Although CD is associated to NHL&#44; no cases of BL have been reported in adult patients with celiac disease&#46; We report a patient with long history of CD that developed intestinal BL&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 52 year-old male with a 20-year history of CD with free-gluten diet compliance was admitted for 10<span class="elsevierStyleHsp" style=""></span>kg weight loss&#44; right lower quadrant discomfort&#44; fever&#44; diarrhea&#44; and hypoesthesia in his left mandibular area over a period of 3 weeks&#46; No significant findings were observed on physical examination&#46; Laboratory data showed an erythrocyte sedimentation rate of 96<span class="elsevierStyleHsp" style=""></span>mm&#47;hour and lactate dehydrogenase &#40;LDH&#41; of 1011<span class="elsevierStyleHsp" style=""></span>U&#47;L &#40;normal value&#58; 236&#8211;460<span class="elsevierStyleHsp" style=""></span>U&#47;L&#41;&#46; The complete blood count&#44; renal function test&#44; liver enzymes and CD antibodies were all within normal limits&#46; The anti-HIV test was negative&#46; Abdominal CT showed an irregular thickening of the terminal ileum that protruded to the cecum with multiple enlarged lymph nodes in the ileocecal region &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>a&#41;&#46; During hospital stay&#44; the patient developed right ptosis and ipsilateral IV cranial nerve palsy without other neurological symptoms&#46; A head MRI and a cerebrospinal fluid sample were normal&#46; Colonoscopy exam showed a 4-cm pseudopolipoid ileal lesion with friable surface that protruded through ileocecal valve&#44; which was biopsied &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; Pathology exam of the mass showed a &#8220;starry sky&#8221; pattern composed of several reactive macrophages with phagocytosis of apoptotic tumor cells and diffuse proliferation of neoplastic cells with medium size nuclei that infiltrated the ileal chorion&#46; Immunohistochemistry stains showed expression of CD20&#44; homogenous expression of BCL6&#44; and lack of expression of BCL2&#46; Ki67&#47;MIB1 staining was homogenously positive in almost all tumor cells&#46; These findings were compatible with BL&#46; Bone marrow biopsy also showed lymphoid neoplastic cells&#46; Chemotherapy was administered with combination of cyclophosphamide&#44; ifosfamide&#44; etoposide&#44; vincristine&#44; doxorubicin&#44; cytarabine&#44; methotrexate&#44; and rituximab&#46; The patient had a complete response and resolution of the IV cranial nerve palsy&#46; After 16 months of follow-up the patient remained disease-free&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">NHL represent 1&#8211;4&#37; of the malignancies arising in the gastrointestinal tract&#44; and stomach is the most common site of involvement&#46; CD is associated with an increased risk either intestinal or extra-intestinal NHL&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> Different studies showed that patients with CD diagnosed after age 50 were more likely to develop NHL&#44; suggesting that a prolonged period of continued gluten ingestion before CD diagnosis has a central role&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">4</span></a> Nevertheless a large case series detected NHL in only 0&#46;4&#37; and small-intestinal cancer in 0&#46;08&#37; of patients with CD&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">5</span></a> Furthermore&#44; in other study only 16&#37; of gastrointestinal NHL were B-cell NHL &#40;mainly MALT lymphoma and diffuse large B cell lymphoma&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">6</span></a> The pathophysiologic mechanism related to the development of lymphoproliferative disorder in patients with CD is poorly understood&#46; The loss of epithelial barrier function&#44; migration of aberrant intestinal immune-cells&#44; and dysfunctional immune surveillance may be involved&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">4</span></a> To the best of our knowledge&#44; there is only one case report of intestinal BL in a pediatric patient with CD while the patient was strictly adhering to a gluten-free diet&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> and a single case of colonic Burkitt-like lymphoma in a 75 year-old male with a 2-year history of CD &#40;1&#41;&#46; BL is an aggressive subtype of B-cell NHL that represents 1&#8211;5&#37; of all NHL in adults and a small percentage of gastrointestinal lymphomas&#46;<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> The median age for incidence of BL in adults is 55 years and the most frequent localization of sporadic BL is the abdomen &#40;60&#8211;80&#37;&#41; and the most frequent symptoms include abdominal pain&#44; weight loss&#44; perforation&#44; obstruction&#44; fever&#44; and high levels of LDH&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Central nervous system involvement was described in 13&#37; of adult patients with BL&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Although infrequent&#44; the presence of cranial neuropathy is associated with this malignancy&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In conclusion&#44; we report an unusual association of intestinal BL in an adult patient with a long history of CD well controlled&#46; The presence of B-symptoms&#44; diarrhea&#44; weight loss and neurological manifestation were the manifestations of BL in this patient&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Author contributions</span><p id="par0025" class="elsevierStylePara elsevierViewall">Saad EJ&#44; Laborie MV and Balderramo D designed the report&#59; Saad EJ&#44; Seculini Pati&#241;o CE and Guanchiale L collected the patient&#39;s clinical data&#59; Saad EJ and Balderramo D analyzed the data and wrote the paper&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Institutional review board statement&#58;</span> The study was reviewed and approved by the Research Department of the Hospital Privado Universitario de C&#243;rdoba&#44; Argentina&#46;</p></span></span>"
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