Cases reported • Intestinal Neoplasms; Cancer of Intestines; Intestinal Cancer

1/163. Natural killer cell lymphoma of the small intestine: diagnosis by flow cytometric immunophenotyping of paracentesis fluid.

Natural killer (NK) cell lymphoma is a mass-forming neoplasm of putative NK cell lineage that typically appears in extranodal locations and has the following immunophenotype: CD2 positive, surface CD3 negative, cytoplasmic CD3 positive, and CD56 positive. We report a case of small-intestinal NK cell lymphoma that was originally diagnosed as an enteropathy-associated T-cell lymphoma based on paraffin immunohistochemistry. However, subsequent flow cytometric immunophenotyping of paracentesis fluid resulted in the correct diagnosis. We describe the case to illustrate the usefulness of this technique, which has not previously been described in such a case.

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ranking = 1
keywords = lymphoma, positive
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2/163. T cell lymphoma involving the graft of a multivisceral organ recipient.

Posttransplant lymphoproliferative disorders are typically of B cell origin, whereas T cell lymphomas have been rarely documented. We present a case of a non-Hodgkin's T cell lymphoma involving the intestinal graft of a multivisceral transplant patient. The patient was a 7-year-old girl who underwent at age 5 a multivisceral transplant secondary to short gut syndrome. Baseline immunosuppressive therapy consisted of FK506, methylprednisone, and mycophenolate mofetil. At 2 years posttransplant she presented with fever, diarrhea, nausea, and vomiting. Multiple endoscopic biopsies revealed a severe intensity, diffuse and focally nodular lymphocytic infiltrate composed predominantly of small, monomorphic lymphoid cells with scattered plasma cells and abundant eosinophils. Immunohistochemically, the majority of the lymphoid cells expressed the pan T cell marker CD3. Southern blot analysis revealed rearrangement of the T cell receptor beta chain gene, with germline configuration of the heavy immunoglobulin chain gene, confirming a clonal T cell genotype. in situ hybridization for Epstein Barr virus revealed rare positive lymphoid cells, that were negative with CD3 by immunohistochemical staining. A detailed clinico-radiological work-up revealed no other sites of involvement by the lymphomatous process. After the diagnosis of posttransplant lymphoproliferative disorder, immunosuppression was reduced with a subsequent partial improvement in the endoscopic appearance of the graft and a focal decrease in the lymphocytic infiltrate seen in the follow-up biopsies. Repeat gene rearrangement studies demonstrated germline configuration of both the T cell receptor beta chain gene and the heavy chain immunoglobulin. gene. To our knowledge, this represents the first description of a T cell lymphoma affecting the intestinal allograft of a multivisceral transplant patient.

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ranking = 1.1426814952711
keywords = lymphoma, positive
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3/163. A case of adenocarcinoma of the small intestine in a Japanese patient with Crohn disease: a report with immunohistochemical and oncogenic analyses.

We report a rare case of crohn disease accompanied by a small-bowel carcinoma that developed in a 54-year-old Japanese man. The ulcerating tumor, which histologically proved to be a poorly differentiated adenocarcinoma and dysplasia surrounding the carcinoma, was located in the diseased ileum. The Ki-67 immunoreactive epithelial cells were increased in regenerative mucosa as compared with values for normal mucosa. The Ki-67- and p53-positive cells were increased in dysplasia and carcinoma as compared with values for regenerative or normal mucosa. In contrast, the p21(WAF1/CIP1) immunoreactive cells were decreased in this order. Intense DCC (deleted in colorectal cancer) expression was constantly shown among normal, regenerative, dysplastic and cancerous tissues. No bcl-2 expression and c-Ki-ras mutations were apparent. In conclusion, enhanced epithelial cell proliferation, p53 overexpression, and decrease of p21(WAF1/CIP1) expression may predispose the small-bowel mucosa to dysplasia and carcinoma development in crohn disease.

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keywords = positive
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4/163. Bowel perforation during chemotherapy for non-hodgkin's lymphoma.

Bowel perforation in patients with primary malignant lymphoma usually occurs at the site of tumor. A 78 year-old man underwent chemotherapy for malignant lymphoma. He presented with abdominal pain. An emergency operation was performed under a diagnosis of panperitonitis. At laparotomy, an anal-side perforation approximately 20 cm from the Treiz ligament was observed. drainage and partial resection of the jejunum was performed. Histopathologic examination demonstrated that there was no characteristic finding of malignant lymphoma around the perforation site in the case. Perforation of the small intestine is one of the most critical complications during the chemotherapy for malignant lymphoma. In cases of chemotherapy for malignant lymphoma, especially systemic administration, we should keep in mind the possibility of perforation of the small intestine. Fortunately, emergency surgery saved the patient presented in this report. Early diagnosis and treatment are important to improve prognosis of bowel perforation in patients with primary malignant lymphoma.

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ranking = 1.428261462243
keywords = lymphoma
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5/163. Spontaneously relapsing clonal, mucosal cytotoxic T-cell lymphoproliferative disorder: case report and review of the literature.

Primary T-cell lymphoma of the gastrointestinal tract is a rare and usually aggressive disorder that may be associated with celiac disease. The authors describe a unique case of a clonal proliferation of CD8+ T cells involving the oral mucosa, ileum, and colon of a 35-year-old man that has regressed spontaneously and recurred numerous times over a 9-year period without treatment. The patient's symptoms were limited to occasional rectal bleeding and recurring painful oral ulcers. Within the intestine, these collections of small T cells induced minimal architectural distortions and did not show extensive epitheliotrophism. polymerase chain reaction and sequencing analyses revealed that the identical T-cell clone has been present for more than 9 years and in different mucosal locations in this patient. This may represent a unique T-cell lymphoproliferative process akin to a mucosal counterpart of lymphomatoid papulosis of the skin.

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keywords = lymphoma
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6/163. Calcification in untreated non-Hodgkin's lymphoma of the jejunum.

A 69-year-old Japanese female was admitted to our hospital due to a 2-month history of vomiting after eating. Examination of the small intestinal tract revealed a tumor with calcification in the inner portion, from the horizontal portion to the ascending portion of the duodenum, and jejunojejunostomy was performed. The pathological findings of the tumor gave a diagnosis of non-Hodgkin's lymphoma, diffuse small cleaved cell (Working Formulation classification), B cell type, of the jejunum. Calcification is rarely found in untreated malignant lymphoma and 15 cases of untreated malignant lymphoma with calcification have been reviewed.

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keywords = lymphoma
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7/163. Pigmented nails and strongyloides stercoralis infestation causing clinical worsening in a patient treated for immunoproliferative small intestinal disease: two unusual observations.

immunoproliferative small intestinal disease (IPSID) is commonly reported from developing countries with poor socioeconomic conditions, hygiene, and high frequency of gastrointestinal infections and infestations. The disease requires anti-malignant chemotherapy in lymphomatous stage. Reported here is a 20-year old man with IPSID lymphoma who responded to anti-malignant chemotherapy initially, but later deteriorated due to strongyloides stercoralis infestation, which was treated successfully with mebendazole. Importance of an early recognition and adequate treatment for gastrointestinal infections and infestations before anti-malignant chemotherapy for this disease is highlighted considering the occurrence of this disease in the developing world. The patient developed alternate brown black and white lines in the finger nails after combination chemotherapy, which has not been reported earlier in this disease; the nail changes disappeared 6 months after the withdrawal of doxorubicin suggesting this drug as the cause for such nail changes during anti-malignant combination chemotherapy.

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keywords = lymphoma
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8/163. Very late onset small intestinal B cell lymphoma associated with primary intestinal lymphangiectasia and diffuse cutaneous warts.

As only a handful of lymphoma cases have been reported in conjunction with primary intestinal lymphangiectasia, it is not yet clear if this association is merely fortuitous or related to primary intestinal lymphangiectasia induced immune deficiency. We report on two female patients, 50 and 58 years old, who developed small intestinal high grade B cell lymphoma a long time (45 and 40 years, respectively) after the initial clinical manifestations of primary intestinal lymphangiectasia. They presented with a longstanding history of fluctuating protein losing enteropathy, multiple cutaneous plane warts, and markedly dilated mucosal and submucosal lymphatic channels in duodenal biopsies. One had a large ulcerated tumour of the proximal ileum and the other diffuse ileal infiltration. In both, histological examination showed centroblastic high grade B cell lymphoma associated with duodenojejuno-ileal mucosal and submucosal lymphangiectasia. They were subsequently successfully treated with surgery and postoperative chemotherapy (AVmCP: adriamycin, cyclophosphamide, Vm26, and prednisolone), and chemotherapy alone (PACOB: adriamycin, cyclophosphamide, vincristine, bleomycine, and prednisolone), respectively. A three year follow up in both cases showed persistent diffuse lymphangiectasia without evidence of lymphoma. The present findings support the hypothesis that primary intestinal lymphangiectasia is associated with lymphoma development.

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keywords = lymphoma
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9/163. Refractory sprue syndrome with clonal intraepithelial lymphocytes evolving into overt enteropathy-type intestinal T-cell lymphoma.

INTRODUCTION: Recently, patients with refractory sprue have been shown to contain a clonal proliferation of phenotypically abnormal intraepithelial lymphocytes in their intestine. Whether this signifies early enteropathy-type intestinal T-cell lymphoma (EITCL) or a reactive condition is not clear. We report on a patient presenting with the findings of refractory sprue who subsequently developed overt EITCL. MATERIAL AND methods: Duodenal biopsies from 1997 (refractory sprue) and duodenal and jejunal biopsies from 1998 (intestinal T-cell lymphoma) were compared by immunohistochemistry and PCR for the detection of T-cell receptor (TCR)-gamma gene rearrangements. Clonal PCR products were sequenced. RESULTS: The duodenal biopsies from both 1997 and 1998 and the jejunal tumor biopsy showed villus atrophy and an increase of intraepithelial lymphocytes with an abnormal immunophenotype (CD3+, CD4-, CD8- and TCR-beta-). In all duodenal specimens including the one from 1997, and the jenunal tumor biopsy, an identical clonal amplificate was detected by enzymatic amplification of the TCR-gamma gene. CONCLUSION: These data suggest that refractory sprue containing a clonal proliferation of phenotypically abnormal intraepithelial lymphocytes may represent an early manifestation of EITCL. The detection of immunohistochemical negativity for several antigens normally found on intraepithelial lymphocytes such as CD8 or the TCR-beta chain in combination with clonal T-cell populations by PCR may be helpful in identifying refractory sprue with a malignant transformation.

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ranking = 0.85695687734582
keywords = lymphoma
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10/163. Successful allogeneic stem-cell transplantation with prophylactic stepwise G-CSF primed-DLIs for relapse after autologous transplantation in mantle cell lymphoma: a case report and literature review on the evidence of GVL effects in MCL.

Mantle cell lymphoma (MCL) is a distinctive clinicopathologic entity and represents 2-8% of all non-Hodgkin's lymphomas. The median survival of patients with MCL is only 3 years, and none of the available conventional chemotherapy regimens appears curative. Encouraging results have been reported with high-dose chemotherapy with autologous stem-cell transplantation (autoSCT). However, a plateau in disease-free survival was not observed in relapsed MCL on the autoSCT trials. Promisingly, alloSCT appears to induce durable remissions via a graft-versus-lymphoma (GVL) effect. Donor lymphocyte infusions (DLIs), by virtue of a GVL effect, have been shown to induce durable remissions in a few cases with refractory MCL that recur after alloSCT. In this article, we review the literature on the evidence of the GVL effects in MCL and describe a patient with relapsed MCL shortly after high-dose chemotherapy with autoSCT. The patient was then successfully treated with Bu/Cy/VP-16 for an alloSCT followed by DLIs in a stepwise fashion. MNCs > 10 x 10(8)/kg were collected by two large-volume leukaphereses from the donor. Harvested stem cells from the 2(nd) day were cryopreserved for the future use as prophylactic DLIs to be given in a stepwise fashion. Cyclosporin and methotrexate were used for GVHD prophylaxis. He had achieved only a partial response by D+64 post transplant. G-CSF-primed cryopreserved DLIs were then infused on D+64 and D+92 to enhance the GVL effect. Grade 3 intestinal GVHD developed 20 days after the 2(nd) DLI and was partially controlled with the combination of cyclosporin, prednisone, and mycophenolate mofetil. Clinical complete remission was observed at D+112, and maintained until the last follow-up day (D+615). Our findings suggest that alloSCT followed by prophylactic DLIs may offer a curative approach to refractory MCL.

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keywords = lymphoma
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