Cases reported "Splenic Neoplasms"

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1/93. Inflammatory pseudotumor of the spleen associated with a cavernous hemangioma diagnosed at intra-operative cytology: report of a case and review of literature.

    This report presents a case of a 40 year old Caucasian female with a 15 cm inflammatory pseudotumor (IPT) of the spleen with associated areas of splenic hemangioma of the cavernous type. Abdominal CT showed a largely fatty splenic mass with enhancing septations, and scattered calcifications, and a small density in the liver. Grossly, the splenic lesion showed a lobulated cut surface with areas of myxoid change, necrosis, hemorrhage and cystic softening. The diagnosis of IPT was suggested at intraoperative consultation using cytologic smears and was, subsequently confirmed on permanent sections. Histologically, the lesion consisted of a densely collagenized spindle cell stroma with patchy aggregates of lymphocytes and plasma cells, and scattered foci showing hemosiderin-laden macrophages extracellular calcium deposits and osseous metaplasia. The stromal spindle cells were immuoreactive for smooth muscle actin and vimentin confirming their myofibroblastic phenotype. There were extensive areas of infarction within the mass. The patient, however, remained asymptomatic preoperatively. Histologic analysis in this case raises the possibility that low grade, perhaps repetitive, trauma to the hemangioma may have resulted in intralesional hemorrhages which, through a process of organization, may have evolved into this sizable inflammatory pseudotumor. In addition, this report reviews the current literature on the clinical significance and presentation, morphologic and immunohistochemical findings, prognosis, differential diagnosis, pathogenesis and therapy of the splenic IPT.
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2/93. Pathologic-spontaneous-rupture of the spleen as a presenting sign of splenic T-cell lymphoma--case report with review.

    A 39 year-old man presented for surgery with epigastric pain, tachycardia, hypotension and a progressive decrease of hemoglobin due to blood loss. Immediate abdominal ultrasonography followed by prompt paracentesis revealed massive intraperitoneal hemorrhage. During emergency laparotomy, a linear, actively bleeding rupture of an enlarged spleen was found and splenectomy was performed. The patient survived and the post-operative course was uneventful. Histopathology of the spleen as well as bone marrow biopsy confirmed the diagnosis of T-Cell lymphoma. Chemotherapy was initiated 3 weeks after surgery. To the best of our knowledge, this is the first reported case of previously undiagnosed T-Cell lymphoma presenting as pathologic rupture of the spleen.
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3/93. Hepatosplenic B-cell lymphoma associated with hemophagocytic syndrome: a case report.

    While T-cell non-Hodgkin's lymphoma (NHL) associated with hemophagocytic syndrome (HPS) has been frequently observed, B-cell NHL associated with HPS has been rarely reported. We report a case of hepatosplenic B-cell lymphoma associated with HPS in a 41-year-old woman who presented with fever of unknown origin. An abdominal CT scan revealed splenomegaly with focal splenic infarction. splenectomy and a liver wedge biopsy showed sinusoidal-pattern infiltration of medium to large tumor cells with positive reaction to a B-lymphocyte marker. Findings on bone marrow examination showed proliferation of histiocytes with avid hemophagocytosis.
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4/93. Hepatosplenic T-cell lymphoma of alphabeta lineage in a 16-year-old boy presenting with hemolytic anemia and thrombocytopenia.

    The authors report an unusual case of peripheral T-cell lymphoma in a 16-year-old boy who presented initially with jaundice, splenomegaly, anemia, and thrombocytopenia. A lymphoma was found subsequently in the spleen, which was infiltrated extensively in the red pulp by medium-sized, blastic-appearing lymphoma cells. Immunologic characterization of these cells revealed positivity for CD3, CD5, CD45RO, CD56, and T-cell intracellular antigen (TIA), and negativity for CD2, CD3, CD4, CD8, CD57, CD34, and terminal deoxynucleotidyl transferase (TdT). Conventional cytogenetic studies revealed the presence of isochromosome 7q. On follow up, this patient deteriorated rapidly, with evidence of liver and bone marrow involvement. Although the overall clinical and pathologic features of this disease were characteristic of hepatosplenic gammadelta T-cell lymphoma, the T-cell receptor of this tumor showed an immunophenotype of alphabeta not gammadelta lineage. Using the Southern blot technique, the authors demonstrated monoclonal gene rearrangement of the T-cell receptor beta-chain. Thus, they confirmed the existence of hepatosplenic alphabeta T-cell lymphoma. In view of its overall similarity to hepatosplenic gammadelta T-cell lymphoma, this unusual entity probably represents a slight biologic variation of the same disease.
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5/93. Blastic transformation of splenic lymphoma with villous lymphocytes after a well-controlled chronic phase of more than 10 years.

    A 30-year-old Japanese man with splenomegaly and lymphocytosis was examined in 1985. blood analysis revealed that some of the lymphocytes had short-surface villi with polar distribution. The cells showed Ig lambda , CD5 , CD11c , CD19 , CD22 , CD23 , CD24 , FMC7 phenotype. A small M peak was detected in the serum. Splenic lymphoma with villous lymphocytes (SLVL) was diagnosed on the basis of these findings. Remission was induced and was maintained with low-dose chlorambucil for more than 10 years. In 1996, the patient developed splenomegaly and lymphadenopathy with "B" symptoms and a high serum lactase dehydrogenase (LDH) level. Large blastoid cells with prominent nucleoli were observed in the bone marrow; later, a small number appeared in the peripheral blood. The bone marrow cells showed a complex chromosomal abnormality involving del(7)(q32). Southern blot analysis of immunoglobulin gene rearrangements in SLVL cells that had been cryopreserved in 1986 and of bone marrow cells in 1996 showed 2 rearranged bands in each cell sample; 1 band showed identical sizes in the 2 samples, and the other showed different sizes. These findings suggest that the blastoid cells were derived from SLVL cells through transformation. After this transformation, the disease followed a highly aggressive course. Various chemotherapeutic agents had little effect, and the patient died 3 months later.
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6/93. Hepatosplenic gamma-delta T-cell lymphoma as a late-onset posttransplant lymphoproliferative disorder in renal transplant recipients.

    We report 2 cases of renal transplant recipients in whom hepatosplenic gamma-delta T-cell lymphoma (gamma-delta HSTCL) developed 5 and 10 years after transplantation. Both patients had marked hepatosplenomegaly, B symptoms (weight loss, fever, and night sweats), and abnormal peripheral blood findings, including anemia in both, thrombocytopenia and leukoerythroblastic changes in 1, and leukocytosis in the other. Markedly atypical lymphoid infiltrate of intermediate to large cells was observed in the spleen, liver, and bone marrow. The malignant cells showed typical immunophenotype of gamma-delta T cells (CD2 , CD3 , CD4-, CD8-, CD7 , gamma-delta T-cell receptor-positive, and alpha-beta T-cell receptor-negative) with clonal T-cell receptor gene rearrangement and were of the V-delta-1 subset. In addition, the cells contained a cytolytic granule-associated protein, TIA-1, and Fas ligand, indicating cytotoxic T-cell differentiation. The malignant T cells in both cases were of host tissue origin. Both cases were negative for Epstein-Barr virus genome using Southern blot analysis. The patients did not respond to reduction of immunosuppression. Despite initial response to chemotherapy, both patients died within 6 months of diagnosis. Our findings indicate that gamma-delta HSTCL can occur as a late complication in transplant recipients.
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7/93. Splenic lymphoma presenting as warm autoimmune hemolytic anemia associated with pure red cell aplasia.

    BACKGROUND AND OBJECTIVE: Warm autoimmune hemolytic anemia (AIHA) is a condition in which peripheral red blood cell (RBC) destruction is induced by the presence of an autoantibody. Pure red cell aplasia (PRCA) represents an isolated process of decreased erythropoiesis. The combination of both is quite rare, with a very poor prognosis. We describe a patient with isolated splenic lymphoma whose presentation was a combination of AIHA and PRCA. The patient was resistant to all treatment. MATERIALS AND methods: Erythroid colony assays were performed, in order to compare the effect of the patient's serum on colonies with that of a normal control. RESULTS: The patient's serum significantly suppressed normal erythroid colony growth. A red cell eluate revealed the presence of a warm autoantibody. CONCLUSIONS: The patient's serum contained warm autoantibody responsible for peripheral RBC destruction and a humoral factor, perhaps the warm autoantibody, which suppressed bone marrow erythropoiesis. Establishing an early diagnosis, and treatment of the underlying disease might result in a better prognosis.
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8/93. Blastic transformation of splenic marginal zone B-cell lymphoma.

    To our knowledge, blastic transformation of splenic marginal zone lymphoma, a recently characterized low-grade lymphoproliferative disorder, has not been reported previously. In this regard, we report the unique case of a 70-year-old woman whose untreated splenic marginal zone lymphoma underwent blastic transformation 3 years after diagnosis. Her hematologic medical history started in 1988 as thrombocytopenia refractory to steroids associated with atypical lymphoid infiltrate in the bone marrow. She underwent splenectomy in 1989, which revealed splenic marginal zone lymphoma. One year later, the patient developed lymphadenopathy noted in the chest, axillary, abdominal, and retroperitoneal lymph nodes. Because she was asymptomatic, treatment was limited to a conservative supportive regimen. The nodal lymphoma cells had features associated with marginal zone lymphoma and expressed B-cell monotypic kappa light chain. She was readmitted for the last time 2 years later with findings of 16% blasts in the peripheral blood and massive infiltration of the bone marrow by large blastoid cells. The blasts showed dispersed chromatin and prominent nucleoli, and possessed a moderate amount of clear cytoplasm. The blasts, like the previous nodal and splenic lymphomas, had a CD20-, CD19-, IgM-positive phenotype, but lacked reactivity for CD5, CD10, and CD23. The patient displayed clinical remission after treatment with vincristine and prednisone, but died of aspiration pneumonia 1 month later. These observations suggest that, similar to the other low-grade lymphoproliferative disorders, an untreated splenic marginal zone lymphoma may undergo high-grade blastic transformation.
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keywords = bone
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9/93. Intravascular occlusive therapy: use of interventional radiology in cancer patients.

    Selective transcatheter intravascular occlusion in the treatment of cancer patients is a valuable extension of interventional diagnostic radiology. Intra-arterial embolization may be performed with various substances, including autologous clot, autologous subcutaneous tissue, Gelfoam, and stainless steel coil. Clinical applications in cancer patients include control of gastrointestinal and genitourinary hemorrhage, preoperative reduction of tumor vascularity, control of local symptoms, and therapeutic reduction of tumor bulk. The technique has been used for preoperative and palliative treatment of neoplasms of the head and neck, kidney, liver, spleen, and soft tissue and bone. Transcatheter intravascular occlusion should be performed only by radiologists experienced in angiographic techniques. Inadvertent occlusion of a normal vessel and thromboembolism are possible complications.
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10/93. Hepatosplenic alphabeta T-cell lymphoma: an unusual case with clinical, histologic, and cytogenetic features of gammadelta hepatosplenic T-cell lymphoma.

    Hepatosplenic gammadelta T-cell lymphoma is a recently identified entity in which lymphoma cells bearing the gammadelta T-cell receptor (TCR) infiltrate the sinusoids of the liver and the sinuses of the splenic red pulp and bone marrow, without lymph node involvement. It is also characterized by a recurrent cytogenetic finding, isochromosome 7q (i7q10). The authors report a case of hepatosplenic lymphoma of alphabeta T-cell phenotype that shares the same clinical, histologic, and cytogenetic characteristics of the previously described hepatosplenic gammadelta T-cell lymphoma. Fluorescent in situ hybridization performed with chromosome 7 probes showed the typical pattern of isochromosome 7q. Genomic analysis of the TCR gamma locus failed to detect a clonal rearrangement. This unique case of hepatosplenic lymphoma of alphabeta T-cell phenotype supports the possibility that lymphoid populations of different alphabeta or gammadelta phenotype that share similar homing and presumably functional properties could give rise to lymphomas displaying similar clinical and pathologic findings.
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