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21/97. radiation therapy and combination of cladribine, cyclophosphamide, and prednisone as treatment of Bing-Neel syndrome: Case report and review of the literature.

    Waldenstrom's macroglobulinemia is a low-grade lymphoma that produces monoclonal IgM. central nervous system symptoms are frequent in Waldenstrom's macroglobulinemia, mostly associated with blood hyperviscosity. Nevertheless, central nervous system infiltration by malignant cells (Bing-Neel syndrome) has rarely been reported. We describe the case of a 72-year-old man with Waldenstrom's macroglobulinemia and central nervous system infiltration by malignant cells with tumor formation. All similar cases reported in the literature are reviewed and the different therapeutic approaches discussed.
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22/97. Monoclonal immunoglobulinaemia in rheumatological practice.

    Monoclonal immunoglobulinaemia is the manifestation of a number of diseases with a quite different prognosis. In some instances in can be associated with articular symptomatology. The authors present an analysis of case-records of four patients where monoclonal immunoglobulinaemia was associated with signs of rheumatic disease--oligoarthritis or polyarthritis of the rheumatoid type. In one instance Waldenstrom's macroglobulinaemia with sjogren's syndrome and hyperviscosity syndrome was involved. In another patient a myeloma was diagnosed. In the remaining two patients a more benign type of monoclonal immunoglobulinaemia was involved.
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23/97. Cast nephropathy in a case of Waldenstrom's macroglobulinemia.

    Waldenstrom's macroglobulinemia is a low-grade lymphoplasmacytic lymphoma characterized by a circulating monoclonal IgM. The clinical manifestations are due to deposition of IgM in liver, spleen, and/or lymph nodes. Related symptoms include anemia and complications of the hyperviscosity syndrome. Renal involvement in classical cases of Waldenstrom's macroglobulinemia is rare, and the pathological hallmark finding in the renal biopsy specimen is a thrombotic microangiopathy. We report the case of a 73-year-old female with the diagnosis of pernicious anemia for 2 years before she presented with acute renal failure. A renal biopsy performed suggested the diagnosis of myeloma cast nephropathy. However, bone marrow biopsy specimens and hematological studies did not support this diagnosis. serum and urinary protein electrophoresis revealed a monoclonal lambda subtype IgM. Ultrasound imaging showed an enlarged spleen. The diagnosis of cast nephropathy in a patient with Waldenstrom's macroglobulinemia was made. She underwent treatment with fludarabine and plasmapheresis/hemodialysis with dramatic improvement of her renal function.
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24/97. Advanced waldenstrom's macroglobulinemia: a case of possible cure after systemic chemotherapy, splenic radiation and splenectomy.

    A 41-year-old man with advanced Waldenstrom's macroglobulinemia (WM) associated with the hyperviscosity syndrome, massive splenomegaly and with IgM concentration of 10 g/dl, was treated in January 1984 with plasmapheresis, systemic chemotherapy (M2 protocol) and splenic radiotherapy. He rapidly improved and was discharged 1 month later. Fourteen months later he underwent splenectomy since a mild splenomegaly persisted though the normalization of bone marrow, peripheral blood and electrophoresis with an IgM concentration of 140 mg/dl. However, at this time immunofixation and immunoelectrophoresis showed a small IgM-kappa monoclonal component. The histological and immunohistochemical analysis showed minimal splenic involvement by WM. Two months after splenectomy, immunofixation and immunoelectrophoresis showed no monoclonal component. The spleen was the probable site of minimal residual disease. The patient was treated with monthly chlorambucil and prednisone for 2 years. Subsequently clinical and laboratory tests persisted within normal limits. The last control performed in January 2002 showed that the patient was in good health; bone marrow examination (aspiration, biopsy with immunohistochemical analysis) and immunofixation persisted normally. This interesting case report, with advanced WM, alive, in good health and without signs of disease 18 years from diagnosis, is presented here and the role of splenectomy is debated.
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25/97. Transfusion-associated graft-versus-host disease in a patient with Waldenstrom's macroglobulinaemia.

    BACKGROUND AND OBJECTIVES: Routine irradiation of cellular blood products is not presently recommended for patients with non-Hodgkin's lymphoma (NHL). MATERIALS AND methods: We report the case of a 72-year-old-man with Waldenstrom's macroglobulinaemia who developed transfusion-associated graft-versus-host disease (TA-GvHD) 13 days following a non-irradiated red cell transfusion. RESULTS: The patient had not previously received purine analogues and none of the donors was homozygous for a human leucocyte antigen (HLA) haplotype that was shared by the recipient. Therefore, his only apparent risk factor was lymphoplasmacytoid NHL. CONCLUSIONS: This case further strengthens the argument that NHL per se is a risk factor for TA-GvHD and supports the proposal that the guidelines for prophylactic irradiation of cellular blood products be extended to include all cases of NHL.
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26/97. No V(H) somatic hypermutation was detected in B-cells of a patient with macroglobulinemia due to splenic marginal zone lymphoma.

    B-cell diseases are classified on the basis of the normal differentiation stages. We report here a case of a patient with a long history of leukocytosis, splenomegaly without lymphadenopathy, and hyperviscosity symptoms. Clinically, the patient's diagnosis was leukemic waldenstrom macroglobulinemia. Chromosomal analysis revealed translocation t(2;7)(p11;q22) along with disease progression. death occurred from pulmonary infection at 46 months after the initial presentation. At autopsy, malignant lymphocytes were found in the marginal areas of the spleen with spreading to the bone marrow and the liver. The histologic findings were consistent with splenic marginal zone lymphoma. We examined the sequences of the immunoglobulin V(H) gene in cells from the initial peripheral blood and from the spleen at autopsy and found that the sequences were identical and had no somatic hypermutation. Macroglobulinemia can occur in various B-cell disorders, including splenic marginal zone lymphoma, even with the transformation of unmutated b-lymphocytes.
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27/97. Myelopathy from Waldenstrom's macroglobulinemia: improvement after Rituximab therapy.

    Approximately 20% of patients with Waldenstrom's macroglobulinemia (WM) have neurological complications; primarily peripheral neuropathies and symptoms related to a hyperviscosity syndrome. We report a rare case of a patient presenting with a slowly progressive myelopathy due to WM who had a marked response to Rituximab therapy.
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28/97. Cutaneous Waldenstrom's macroglobulinaemia.

    We report a case of Waldenstrom's macroglobulinaemia that presented with infiltrated skin nodules and plaques but without systemic symptoms. Cutaneous manifestations such as purpura, oedema, urticaria and ulceration may be seen in Waldenstrom's macroglobulinaemia and are a consequence of hyperviscosity, cryoglobulinaemia and/or tissue deposition of immunoglobulins. Direct cutaneous infiltration by neoplastic lymphoid cells is less common, and very rare as the initial presentation of Waldenstrom's macroglobulinaemia.
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29/97. Waldenstrom's macrogloblinaemia: modified plasmapheresis as treatment option in a nigeria setting.

    We describe a case of Waldenstroms macroglobulinaemia in a 56 year old Negroid male, to whom modified plasmapheresis/plasma exchange was offered with good control of symptomatic hyperviscosity Amelioration of the clinical status and objective assessment of response was evident by dramatic reduction in monoclonal immunoglobulins M (IgM) from 100g/L to 14.7 g/L and a fall in erythrocyte sedimentation rate from 130 mm/hr to 80 mm/hr with this therapy. This highlights the need to adopt a modification of manual plasmapheresis in the treatment of the hyperviscosity syndrome complicating this B-cell disease in this environment.
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30/97. Coincidence of primary myelodysplastic syndrome and non-Hodgkin lymphoma.

    A 60-year-old patient is presented with primary myelodysplastic syndrome classified according to the criteria of the French-American-British classification as chronic myelomonocytic leukemia and with a hyperviscosity syndrome. We found a monoclonal gammopathy with a very high serum immunoglobulin M level. There is strong evidence for coincidence of myelodysplastic syndrome and Waldenstrom's macroglobulinemia in this patient.
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