Cytotoxic Chemotherapy Successfully Induces Durable Complete Remission in 2 Patients with Mosquito Allergy Resulting from Epstein-Barr Virus-Associated T-/Natural Killer Cell Lymphoproliferative Disease

Koyama, M.; Takeshita, Yasufumi; Sakata, Akifumi; Sawada, Akihisa; Yasui, Masayoshi; Okamura, Takayuki; Inoue, Masami; Kawa, Keisei

doi:10.1532/ijh97.05083

Cited by 24 publications

(18 citation statements)

References 10 publications

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“…Thus far, two patients with hypersensitivity to mosquito bites (1 NK type and 1 gdT type) who received HDCA have remained in CR for 46 years. 19 In addition, we have succeeded to treat one more patient who developed EBV-infected T-cell LPD after cardiac transplant (sustained CR for 42 years). Therefore, more effective treatment strategy, including new combination chemotherapy as well as other approaches, remains to be established.…”

Section: Discussionmentioning

confidence: 99%

Excellent outcome of allogeneic hematopoietic SCT with reduced-intensity conditioning for the treatment of chronic active EBV infection

Kawa

Sawada

Sato

et al. 2010

Bone Marrow Transplant

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Since we reported the first successful case of allogeneic hematopoietic SCT (allo-HSCT), we have performed allo-HSCT for 29 patients with chronic active EBV infection (CAEBV), using either myeloablative conditioning (MAC) allo-HSCT (MAST) or reduced-intensity conditioning (RIC) allo-HSCT (RIST). In this retrospective analysis we compared the outcomes after MAST and RIST to identify the optimal conditioning for patients with CAEBV. Of 29 patients, 11 underwent allo-HSCT with MAC, consisting of TBI (12 Gy), etoposide (900 mg/ m 2 ) and CY (120 mg/kg) or melphalan (210 mg/m 2 ), and the remaining 18 patients received allo-HSCT after RIC, consisting of fludarabine (B180 mg/m 2 ) and melphalan (140 mg/m 2 ) or CY (120 mg/kg), with/without antithymocyte globulin and low-dose irradiation. Donor sources were 8 related BM, 2 related peripheral blood, 5 CD34 selected cells from HLA-haploidentical donors, 8 unrelated BM and 8 unrelated cord blood. The 3-year-EFS rate was 54.5±15.0% for MAST group and 85.0±8.0% for RIST group, and the 3-year OS rate was 54.5 ± 15.0% for MAST group and 95.0 ± 4.9% for RIST group (P ¼ 0.016). Allo-HSCT after RIC seems to be a promising approach for the treatment of CAEBV.

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Section: Discussionmentioning

confidence: 99%

Excellent outcome of allogeneic hematopoietic SCT with reduced-intensity conditioning for the treatment of chronic active EBV infection

Kawa

Sawada

Sato

et al. 2010

Bone Marrow Transplant

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“…The optimal strategy for the treatment of CAEBV is controversial. The administration of chemotherapies, immunosuppressant therapies, recombinant interleukin-2, interferon-alpha, or antiviral agents seems to lead to complete virus elimination in some cases [19][20][21], but the reports of these successes have been somewhat vague. In the meantime, allo SCT has been suggested as a curative therapy for CAEBV, especially in adult cases [11][12][13][14].…”

Section: Discussionmentioning

confidence: 99%

Severe degenerative change of multiple organs mediated by chronic active Epstein–Barr virus infection with infected T-cell expansion

et al. 2008

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We here report the case of a young Japanese woman diagnosed with chronic active Epstein-Barr virus (EBV) infection. Intensive therapy with the CHOP regimen was partially able to control virus expansion, but various central nervous system symptoms appeared and gradually progressed. EBV-encoded RNA, detected using in situ hybridization, disclosed the presence of EBV in liver and bone marrow tissue, and real-time PCR revealed the presence of EBV in the cerebrospinal fluid (CSF) and serum. CD3+CD4+CD8-CD56- T-cell expansion in the peripheral blood (PB) and CSF was also observed. Atrophic brain changes were progressive, and the patient died of central nervous system disturbance and pulmonary hemorrhage a year after diagnosis. Autopsy revealed that EBV-infected T lymphocytes with a phenotype similar to those seen in PB and CSF had infiltrated multiple organs: the lymph nodes, bone marrow, endocardium, pericardium, myocardium, spleen, liver, and spinal cord. There have been few previous reports of severe degenerative changes in the myocardium, liver, and spinal cord in patients with EBV infection. Although EBV occasionally infiltrates the central nervous system and brain, atrophic changes mediated by EBV are rare. The autopsy results of this case suggest the possibility of EBV-mediated, severe degenerative changes in multiple organs.

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“…Koyama and colleagues recently reported 2 pediatric patients with EBV-positive T-/NK-cell lymphoproliferative disorders and HMB who were successfully treated with sequential chemotherapy consisting of cyclosporine A and prednisolone (CP), followed by CHOP, Capizzi, and HDCA regimens [6], as described below:…”

Section: Treatment Protocolmentioning

confidence: 99%

Clinical features of adult-onset chronic active Epstein–Barr virus infection: a retrospective analysis

et al. 2011

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We performed a retrospective analysis of patients with adult-onset chronic active Epstein-Barr virus infection (CAEBV). First, we analyzed five patients (aged 28-72) diagnosed at our hospitals with EBV-infected clonally proliferating T cells. Four patients were administered cyclophosphamide/doxorubicin/vincristine/prednisone (CHOP) chemotherapy, but no remarkable decrease of viral load was observed in three of the patients. The other patient died 19 days after initiation of CHOP treatment due to disease progression. Addition of high-dose cytarabine to the regimens of two of the patients was discontinued shortly after administration, due to the development of grade 4 pericardial effusion. Together, these regimens may be insufficient for treating adult-onset CAEBV. We next reviewed 23 adult-onset CAEBV patients, adding 18 previously reported patients to the five patients described in the present study. T cells were frequently infected (87%), whereas NK- and T-cell types are known to be almost equally prevalent in childhood-onset cases. The time duration from the onset of disease to initiation of treatment averaged 20 months. Reports showed that 12 patients died; seven patients died at an average of 8 months after initiation of treatment. Patients' disease courses seemed to be rapidly progressive and more aggressive than those of childhood-onset cases. More cases must be studied to clarify clinical features and establish an optimal treatment strategy.

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Cytotoxic Chemotherapy Successfully Induces Durable Complete Remission in 2 Patients with Mosquito Allergy Resulting from Epstein-Barr Virus-Associated T-/Natural Killer Cell Lymphoproliferative Disease

Cited by 24 publications

References 10 publications

Excellent outcome of allogeneic hematopoietic SCT with reduced-intensity conditioning for the treatment of chronic active EBV infection

Excellent outcome of allogeneic hematopoietic SCT with reduced-intensity conditioning for the treatment of chronic active EBV infection

Severe degenerative change of multiple organs mediated by chronic active Epstein–Barr virus infection with infected T-cell expansion

Clinical features of adult-onset chronic active Epstein–Barr virus infection: a retrospective analysis

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