Cases reported "Hypersensitivity"

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1/29. Characterization of Epstein-Barr virus (EBV)-infected natural killer (NK) cell proliferation in patients with severe mosquito allergy; establishment of an IL-2-dependent NK-like cell line.

    The clinical evidence of a relationship between severe hypersensitivity to mosquito bite (HMB) and clonal expansion of EBV-infected NK cells has been accumulated. In order to clarify the mechanism of EBV-induced NK cell proliferation and its relationship with high incidence of leukaemias or lymphomas in HMB patients, we studied clonally expanded NK cells from three HMB patients and succeeded in establishing an EBV-infected NK-like cell line designated KAI3. immunoblotting and reverse transcriptase-polymerase chain reaction (RT-PCR) analyses revealed that KAI3 cells as well as infected NK cells exhibited an EBV latent infection type II, where EBV gene expression was limited to EBNA 1 and LMP1. As KAI3 was established by culture with IL-2, IL-2 responsiveness of peripheral blood NK cells from patients was examined. The results represented markedly augmented IL-2-induced IL-2R alpha expression in NK cells. This characteristic property may contribute to the persistent expansion of infected NK cells. However, KAI3 cells as well as the NK cells from patients were not protected from apoptosis induced by either an anti-Fas antibody or NK-sensitive k562 cells. Preserved sensitivity to apoptosis might explain the relatively regulated NK cell numbers in the peripheral blood of the patients. To our knowledge, KAI3 is the first reported NK-like cell line established from patients of severe chronic active EBV infection (SCAEBV) before the onset of leukaemias or lymphomas. KAI3 cells will contribute to the study of EBV persistency in the NK cell environment and its relationship with high incidence of leukaemias or lymphomas in HMB patients.
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2/29. An adult patient with hypersensitivity to mosquito bites developing mantle cell lymphoma.

    hypersensitivity to mosquito bites (HMB) has been known to occur exclusively in the first 2 decades of life and is frequently associated with Epstein-Barr virus (EBV) infection and lymphoproliferative diseases. We report here the first adult patient with HMB, a 61-year-old Japanese man who developed mantle cell lymphoma. EBV was detected in the lymph node by polymerase chain reaction and by in situ hybridization. serum levels of interleukin (IL)-4, IL-6, and IL-10 were markedly increased, and the T-helper cell (Th)1/Th2 balance determined by intracellular cytokine levels was polarized to Th2. These findings suggest that the Th1/Th2 imbalance could partly be involved in the pathogenesis of HMB.
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3/29. hypersensitivity to mosquito bites is not an allergic disease, but an Epstein-Barr virus-associated lymphoproliferative disease.

    Recently, we showed that 5 cases of hypersensitivity to mosquito bites (HMB) concealed the clonal lymphoproliferation of Epstein-Barr viral (EBV) dna-positive natural killer (NK) cells. Although the symptoms of HMB have been supposed to derive from Arthus phenomenon, it has become apparent that this unique disorder has the potential to develop into so-called malignant histiocytosis (MH) or related disorders. Accordingly, the criteria for MH have been changed, and a newer diagnostic name, hemophagocytic syndrome, has been described as being associated with viral infection or leukemia/lymphoma. We previously reported that biopsy specimens taken from skin lesions demonstrated infiltration of lymphocytes bearing the phenotype of NK cells. In this study, we found that skin lesions exhibited infiltration of atypical lymphocytes around the small vessels, resembling angiocentric lymphoma, and that these infiltrating cells were positive for EBER-1 by in situ hybridization. These findings support the concept that HMB is the most important manifestation of a certain type of lymphoproliferative disease that presents with an intense local skin reaction and high fever following mosquito bites, and whose essence is the lymphoproliferation of EBV dna-positive NK cells.
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4/29. hypersensitivity to mosquito bites with natural-killer cell lymphocytosis: the possible implication of Epstein-Barr virus reactivation.

    hypersensitivity to mosquito bites (HMB) has been recognized as one of the wide variety of Epstein-Barr virus (EBV)-associated lymphoproliferative disorders, some of which overlap with chronic active EBV infection and hydroa vacciniforme. It frequently predisposes to hemophagocytic syndrome or lymphoid malignancy with a clonal EBV replication. Here we report an 8-year-old Japanese boy who presented with a 3-year history of HMB. Every summer, he had repeated severe local skin reactions with high-grade fever, lymphadenopathy, and mild liver dysfunction after mosquito bites. Histologically, lymphoid cells overexpressing EBV-encoded small nuclear RNAs (EBER), a marker of latent EBV infection, and eosinophils infiltrated diffusely into the lesional skin. His blood tests showed a marked increase in the CD11b 16 56 natural-killer cell population (~ 50% of the peripheral lymphocytes), but neither a clonal proliferation of EBV nor abnormal antibody titers to EBV. Quantitative PCR using a cloned EBV-determined nuclear antigen (EBNA)-1 vector revealed that a high copy number of the EBV genome is integrated into his peripheral blood cells even during the mosquito-free winter. Based on this evidence, he was diagnosed as being in a smoldering stage of HMB, in which EBV-associated NK cell lymphoproliferation might participate in its pathogenesis.
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5/29. A possible mechanism of NK cell-lineage granular lymphocyte proliferative disorder (NK-GLPD) in a patient with chronic active Epstein-Barr virus infection (CAEBV) and severe hypersensitivity to mosquito bites (SHMB).

    We report the case of a young female patient with chronic active Epstein-Barr virus infection (CAEBV) and severe hypersensitivity to mosquito bites (SHMB). She showed a marked increase of NK cell population in peripheral blood. The NK cell population was suggested to be infected with EBV, and to be oligoclonal by Southern blotting using an EBV genome terminal-repeat probe. The NK cells aberrantly expressed CD25, a high affinity receptor for IL-2, and showed an augmented in vitro proliferative response to IL-2. Moreover, they also showed enhanced expression of both Fas-ligand and Bcl-2, and resistance to in vitro Fas-induced apoptotic cell death (Fas-ACD). Taken together, these observations suggested that both the augmentation of proliferative response to IL-2 and the decrease in Fas-ACD may cause NK cell lineage granular lymphocyte proliferative disorder (NK-GLPD) in patients with CAEBV and SHMB.
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6/29. hypersensitivity to mosquito bites associated with natural killer cell-derived large granular lymphocyte lymphocytosis: a case report in korea.

    hypersensitivity to mosquito bites (HMB) is characterized by intense skin reactions at bite sites. The pathogenesis of HMB might be related to clonal lymphoproliferation of Epstein-Barr virus dna-positive natural killer (NK) cells. We report the first case of HMB possibly associated with NK cell-derived large granular lymphocyte (NK-LGL) lymphocytosis in korea.
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7/29. Circulating immune complexes and activation of the complement sequence in acute allergic bronchopulmonary aspergillosis.

    A 13-yr-old asthmatic girl with allergic bronchopulmonary aspergillosis (ABPA) exhibited eosinophilia, IgE hypergammaglobulinemia, serum precipitins to Aspergillus, a dual skin reaction to aspergillus fumigatus, and positive, lymphocyte transformation in vitro in response to stimulation with aspergillus fumigatus antigen. The acute phase of the disease was characterized by an increase in blood eosinophilia, a rise in serum IgE, circulating immune complexes, and evidence of activation of the complement system through the classical pathway.
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8/29. CD4 T-lymphocyte-induced Epstein-Barr virus reactivation in a patient with severe hypersensitivity to mosquito bites and Epstein-Barr virus-infected NK cell lymphocytosis.

    BACKGROUND: Natural killer (NK) cell lymphocytosis associated with Epstein-Barr virus (EBV) infection often shows severe hypersensitivity to mosquito bites (HMB) characterized by intense local skin reactions and systemic symptoms such as high fever, lymphadenopathy, and hepatosplenomegaly. However, the induction mechanism of HMB is still unclear. OBSERVATIONS: We investigated a typical case of HMB with EBV-positive NK cell lymphocytosis. CD4 T cells dominantly infiltrated the site of the mosquito bite, while EBV-positive cells were few in comparison. CD4 T cells, but not CD8 T cells or NK cells, responded to the mosquito salivary gland extracts. Interestingly, coculturing of the NK cells and CD4 T cells activated by mosquito extracts induced expression of EBV lytic-cycle proteins in the NK cells. Furthermore, the expression of BZLF1, a viral lytic-cycle transactivator, was detectable at the skin lesion induced by scratch patch testing with mosquito extract. The EBV dna copy number levels in the plasma were elevated in systemic HMB symptoms compared with the normal condition. CONCLUSIONS: CD4 T cells are important for the primary skin reaction to mosquito bites and might play a key role in reactivation of latent EBV infection in NK cells. This viral reactivation contributed to the pathogenesis of the infectious mononucleosis-like systemic symptoms of HMB in our present case.
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9/29. A case of a long-time survivor with chronic active Epstein-Barr virus infection.

    Epstein-Barr virus (EBV) is associated with hypersensitivity to mosquito bites (HMB) and fatal EBV-associated hemophagocytic syndrome (HPS). The prognosis of patients with chronic active EBV infection (CAEBV) is very poor. We report a rare case of an adult woman patient with a 28-yr history of HMB, who developed EBV-HPS. EBV genome was detected in the serum and peripheral blood lymphocytes. Clonal proliferation of EBV was demonstrated by Southern blot analysis using an EBV genome terminal-repeat probe. This is a very rare case of a long-term survivor with CAEBV. The patient was initially treated with immunochemotherapy and achieved complete remission. However, the patient immediately relapsed and underwent allogeneic bone marrow transplantation (BMT) from her HLA-matched brother. Peripheral blood cell recovered well, and EBV genome disappeared from the peripheral blood. Allogeneic BMT may be effective in eradicating EBV-HPS. Unfortunately, the patient died of graft vs. host disease on the 92nd day after BMT.
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10/29. A patient with severe black fly (simuliidae) hypersensitivity referred for evaluation of suspected immunodeficiency.

    BACKGROUND: Biting flies of the diptera order and specifically the black fly (simuliidae family) can be rare causes of severe hypersensitivity reactions. OBJECTIVE: To describe a patient referred for evaluation of immunodeficiency whose clinical course is explained by severe simuliidae hypersensitivity. methods: The patient's immune system was investigated using standard laboratory evaluations. hypersensitivity to simuliidae was pursued because of historical features of her presentation and was specifically examined by skin prick and intradermal testing with whole body extract. RESULTS: The patient's history was notable for recurrent and severe seasonal episodes of presumed cellulitis after black fly bites that responded poorly to intravenous antibiotics. One episode was followed by acute inflammatory demyelinating polyneuropathy (guillain-barre syndrome) and another by minimal change nephrotic syndrome. The results of immunologic investigations were unremarkable, but cutaneous hypersensitivity to simuliidae was demonstrated with a 6.5-mm wheel and 35-mm flare reaction to intradermal injection of only 0.0005 microg of whole body extract. CONCLUSION: Similar to hymenoptera, simuliidae can rarely result in extreme hypersensitivity and should be considered in appropriate cases. This patient illustrates how severe reactions toinsect bites can be misdiagnosed.
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