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1/62. CD4/CD8 double-positive adult T cell leukemia with preceding cytomegaloviral gastroenterocolitis.

    We present a rare case of adult T cell leukemia (ATL) in which leukemic T cells simultaneously expressed CD4 and CD8 surface antigens and refractory cytomegalovirus (CMV)-induced gastroenterocolitis preceded its clinical onset. A 40-year-old male was admitted to our hospital with abdominal pain and bloody stool. biopsy specimens of the gastric and rectal mucosa indicated CMV-induced gastroenterocolitis. The patient also proved to be seropositive for human T lymphotropic virus type I (HTLV-I). While being administered gancyclovir for CMV infection, he presented hepatomegaly and systemic lymphadenopathy. Monoclonal expansion of lymphoid cells integrated with HTLV-I genome was observed. He underwent a LSG15 regimen and hepatomegaly and lymphadenopathy improved markedly. Gastroenterocolitis also improved, but the symptoms did not disappear completely. CMV-induced diseases are prevalent among immunosuppressed patients. Although there was no evidence that this patient had ATL on admission, it is likely that he was severely immunodeficient. CMV can easily infect damaged mucosa. ATL cells often infiltrate gastrointestinal mucosa and may have triggered CMV gastroenterocolitis in this case.
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2/62. Posttransplantation B lymphoblastic leukemia with Burkitt-like features.

    BACKGROUND: Posttransplantation Epstein-Barr virus-associated lymphoproliferative disease (PTLPD) occurs as a spectrum of disease ranging from benign, polyclonal, localized lymphoid hyperplasia to malignant, monoclonal, disseminated lymphoma, sometimes involving the bone marrow. To our knowledge, PTLPD has not been previously reported to present as acute lymphoblastic leukemia. methods: We report the case of a boy who developed PTLPD in the form of acute lymphoblastic leukemia 6 years after cardiac transplantation. He had greater than 90% bone marrow invasion by Epstein-Barr virus-positive B lymphoblasts with Burkitt-like features and a t(8;14) translocation. RESULTS: He was successfully treated with combination chemotherapy but unfortunately died, 6 months after completing treatment, from ischemic heart disease. CONCLUSIONS: B lymphoblastic leukemia may occur as a manifestation of PTLPD and should be included in the classification of these diseases. bone marrow examination should be an essential part of the investigation of patients suspected of having PTLPD.
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3/62. Successful treatment of cytomegalovirus colitis with ganciclovir in a patient with adult T cell leukemia lymphoma: case report.

    An 84-year-old patient with adult T cell leukemia lymphoma (ATLL) developed diarrhea on day 5 of chemotherapy and was diagnosed with cytomegalovirus (CMV) colitis. sigmoidoscopy revealed multiple superficial erosions surrounded by a flare. Computed tomography (CT) and ultrasonogram of the abdomen revealed marked thickening of the colonic mucosa. There were 186 CMV antigen-positive leukocytes per 31,000 white blood cells (WBC). A colonic biopsy specimen showed typical CMV nuclear inclusions. Immunohistological study of the specimen was positive for CMV antigen. Administration of ganciclovir (DHPG) 500 mg/day for 14 days improved the diarrhea and other symptoms. On day 30 of the chemotherapy, the patient developed diarrhea again but was diagnosed with pseudomembranous colitis instead of CMV colitis. At that time, CMV antigenemia and a histologic study for CMV were negative. The stool was positive for clostridium difficile toxin antigen. ATLL patients are believed to be immunocompromised hosts and often develop opportunistic infections such as CMV infection. Most suffer from CMV pneumonia at the end of their course of therapy. Few gastrointestinal (GI) CMV infections are seen in ATLL patients and details of CMV colitis have never been reported. When an ATLL patient develops diarrhea that barely responds to conventional therapy, CMV colitis and pseudomembranous colitis should be listed in the differential diagnosis.
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4/62. Fatal herpesvirus-6 encephalitis in a recipient of a T-cell-depleted peripheral blood stem cell transplant from a 3-loci mismatched related donor.

    Human herpesvirus-6 (HHV-6), like all the other herpes viruses, remains latent in host cells after primary infection but can be reactivated in immunocompromised patients causing fever, skin rash, bone marrow (BM) suppression, pneumonitis, sinusitis and meningoencephalitis. We describe the case of a man with chronic myelogenous leukemia who developed encephalitis associated with acute graft-versus-host disease two months after a T-cell-depleted mismatched peripheral blood stem cell transplant. Magnetic resonance images of the brain revealed multiple bilateral foci of signal abnormality. HHV-6 was the only pathogen detected in cerebrospinal fluid by PCR. Treatment with both ganciclovir and foscarnet was unsuccessful and the patient gradually deteriorated and died. Other cases of HHV-6 encephalitis after bone marrow transplantation are reviewed.
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5/62. A case of adult T-cell lymphoma leukemia with hemophagocytic syndrome.

    A 62-year-old Japanese woman was admitted to our clinic with virus-associated hemophagocytic syndrome (VAHS) in subcutaneous adult T-cell lymphoma leukemia (ATLL). Bone marrow aspiration showed hypocellularity, histiocytic hyperplasia, and hemophagocytosis. There was serological evidence of chronic cytomegalovirus (CMV) infection. The hemophagocytic syndrome (HPS) initially improved by some treatments, and the patient later experienced remission several times, but the CMV infection persisted. Most cases of non-tumorous HPS in adults are associated with viral or bacterial infection, and underlying diseases in non-tumorous HPS are mostly blood diseases, especially T-cell lymphoma (1, 2), but ATLL is a rare underlying disease in such cases.
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6/62. The pathology of the temporal bones of a child with acquired cytomegalovirus infection: studies by light microscopy, immunohistochemistry and polymerase-chain reaction.

    STUDY DESIGN: The first case of an acquired cytomegalovirus (CMV) infection of the inner ear is reported in a 3-year-old girl in remission from acute lymphocytic leukemia. methods: Horizontal sections of the temporal bones were studied by light microscopy and immunohistological staining by avidin-biotin-complex-technique was performed on selected archival sections. Three sections were processed for detection of the virus genome by the polymerase chain reaction (PCR). RESULTS: By light microscopy the epithelium of the endolymphatic sac, the utricle and the semicircular canals showed deeply stained acidophilic inclusions and the stria vascularis had a loose structure especially in the intermediate layer. The changes were limited to the non-sensory parts of the labyrinth and no CMV type cells were observed in the organ of corti. There was a loss of inner and outer hair cells and loss of cochlear ganglion cells caused by either the virus or treatment with gentamicin. Standard immunohistochemistry failed to demonstrate staining with CMV antibodies, but PCR, demonstrated CMV-dna in one section. CONCLUSION: Molecular techniques may be able to detect acquired CMV infections in archival pediatric bones temporal bones. The histologic findings in the labyrinth were milder, however showed some similarity to children with congenital CMV labyrinthitis.
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7/62. Hemolytic uremic syndrome in a child with leukemia and cytomegalovirus infection.

    A 2-year-old boy had a severe cytomegalovirus (CMV) infection with multi-organ involvement, while on maintenance therapy for acute lymphoblastic leukemia. The patient was treated with intravenous gancyclovir, with a marked improvement in his clinical status, with the exception of a progressive deterioration of the renal function. He also developed hemolytic anemia and thrombocytopenia, suggesting a diagnosis of atypical hemolytic uremic syndrome (HUS). A percutaneous renal biopsy showed lesions consistent with HUS, but no evidence of CMV infection. The patient had a good clinical outcome with no evidence of renal sequelae. We report a rare association of CMV infection and HUS in the pediatric age-group, which suggests a possible cause-effect relationship that deserves further evaluation.
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8/62. CD8-depleted donor leukocyte transfusions for cytomegalovirus antigenemia in patient with acute lymphoblastic leukemia treated with allogeneic CD34( ) cell transplantation.

    A 24-year-old man with acute lymphoblastic leukemia received an allogeneic CD34( ) cell transplant from an HLA-mismatched sibling because of refractory disease. The CD34( ) cells were enriched by the immunomagnetic method. One month after the transplant his situation became complicated due to cytomegalovirus (CMV) antigenemia, which was resistant to treatment with ganciclovir. He was treated with CD8( ) cell-depleted donor lymphocyte transfusions (CD8-depleted DLT). After CD8-depleted DLT, the CMV antigenemia disappeared completely. This case report suggested that CD8-depleted DLT was an effective therapy for CMV antigenemia after allogeneic CD34( ) cell transplantation.
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9/62. rhodococcus equi and cytomegalovirus pneumonia in a renal transplant patient: diagnosis by fine-needle aspiration biopsy.

    rhodococcus equi is a common cause of pneumonia in animals. Human infection is rare. Increasing number of cases are being reported in immunosuppressed individuals mostly associated with hiv infection, but also in solid organ transplant recipients and leukemia/lymphoma patients. We report on an adult male who developed pneumonia and gastroenteritis 4 mo after receiving a renal transplant. CT scan of the lungs showed a dominant 2.5-cm upper lobe lung mass and smaller bilateral nodules. He underwent a diagnostic bronchoscopy with fine-needle aspiration biopsy of the largest lung nodule. Smears showed histiocytic granulomatous inflammation, foamy macrophages, and acute inflammatory exudate. Scattered foamy macrophages displayed intracellular coccobacilli identifiable on Diff-Quik stain. A few cells with changes suggestive of viral inclusions were identified. cytomegalovirus (CMV) immunostain was positive in the cell block sections. lung cultures grew R. equi. To the best of our knowledge, this is the first report of coinfection with R. equi and CMV.
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10/62. Recurrent disseminated herpes zoster and cytomegalic perianal ulcer: a case report and review of the literature.

    We describe a patient with lymphocytic leukemia who developed multiple, disseminated, vesiculopustular eruptions in combination with perianal ulcer. Four years earlier, she had a herpes zoster (HZ) infection involving the ophthalmic division of her left trigeminal nerve with subsequent postherpetic neuralgia that was treated with steroids. After the studies, we concluded that the patient had a recurrent disseminated HZ infection and perianal ulcer caused by cytomegalovirus (CMV).
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