Hemophagocytic lymphohistiocytosis (HLH) is a rare condition with high mortality. We report a case of a 74-year-old woman with rheumatoid arthritis who developed HLH secondary to pyelonephritis due to Escherichia coli infection following infliximab treatment. Bone marrow aspiration showed proliferation of histiocytes with hemophagocytosis. The patient died despite treatment with intravenous antibiotics intravenous methylprednisolone and intravenous immunoglobulin. Cytokine levels were measured and are discussed.
We present a case of subacute meningoencephalitis associated with human T-lymphotrophic virus Type I (HTLV-I) infection, for HTLV-I antibody being positive in serum and cerebrospinal fluid. Necropsy findings disclosed leptomeningeal and parenchymal mononuclear cell infiltration with multinucleate giant cells, which are similar to those seen in subacute encephalitis with acquired immunodeficiency syndrome (AIDS).
anti-white matter antibodies that mainly recognized astrocytes. Intravenous steroid followed by oral steroid reduced the symptoms to a remarkable degree. The patient has now been successfully sustained with steroid for more than two years. Weconsidered that this case is classified as non-paraneoplastic limbic encephalitis, and acquired autoimmunity played a major role in the pathogenesis of this case. (Internal Medicine 42: 428-432, 2003)
Serum soluble interleukin-2 receptor (sIL-2R) levels were determined in patients with chronic myeloproliferative disorders (CMPD): 18 with chronic myelogenous leukemia in chronic phase (CML in CP), seven with CML in accelerated phase (AP) or blastic crisis (BC), six with polycythemia vera (PV), eight with essential thrombocythemia (ET), one with primary myelofibrosis (PMF), and 50 controls. The mean (+/-S.E.M.) levels were higher in CMPD than in controls (CML in AP or BC, 2693 +/- 694 U/ml, P < 0.0001; CML in CP, 792 +/- 63 U/ml, P < 0.0001; PV 553 +/- 89 U/ml, P < 0.05; ET, 449 +/- 56 U/ml; PMF, 628 U/ml vs. controls, 395 +/- 25 U/ml). Patients with CML in CP had significantly higher serum sIL-2R levels than patients with ET (P < 0.005), and levels were markedly elevated in AP and BC (P < 0.001). Serum sIL-2R levels were positively correlated with WBC count and lactic dehydrogenase in CMPD, and in CML in CP. Serum sIL-2R levels in CMPD were negatively correlated with RBC and platelet counts. Serum sIL-2R levels were significantly lower in patients with CML in CP who showed a cytogenetic response after interferon (IFN) therapy than in those who showed no response (P < 0.05). These findings suggest that a high serum sIL-2R level reflects the leukocyte growth in CMPD and is useful both for differentiating CML from other CMPD and for predicting the response to IFN therapy in CML.
An 85-year-old man presented with acute transverse myelopathy: flaccid paraplegia, superficial and deep sensory disturbance below Th6 and loss of urinary sensation. Spinal magnetic resonance image showed an epidural mass compressing the spinal cord at the level of C7 to Th2. Immunoelectrophoresis revealed the presence of immunoglobulin D (IgD)-l M protein. Chemotherapy markedly improved both the haematologic aspect and tumor size, but not the motor deficit. In IgD myeloma, extraosseous spreads occur frequently, but extraosseous epidural tumors causing compression myelopathy are relatively rare. To our knowledge, this is the fourth report of cases.
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