Relapsing Campylobacter coli bacteremia with reactive arthritis in a patient with X-linked agammaglobulinemia.

A patient genetically diagnosed with X-linked agammaglobulinemia repeatedly developed bacteremia due to Campylobacter coli (C. coli) for one year and seven months in spite of immunoglobulin replacement therapy. Throughout the clinical course, C. coli with identical genetic patterns was repeatedly isolated from both blood and stool cultures, thus indicating that the patient had latent intestinal infection. The bacteremia was always accompanied by reactive arthritis. Since the immunoglobulin level was extremely low with severe B cell deficiency, the reactive arthritis must have been induced in a humoral immunity-independent manner. Adding oral minocycline following intravenous meropenem was very effective; the stool cultures became negative and the patient has been well for more than one year without relapse of bacteremia.

[Pure red cell aplasia successfully treated with rituximab in a patient with relapsed diffuse large B-cell lymphoma].

Malignant lymphoma is often accompanied by an autoimmune disorder. Here, we describe a 82-year-old woman with pure red cell aplasia (PRCA) complicated with relapsed diffuse large B-cell lymphoma. Primary treatment involving chemotherapy, continuous oral administration of prednisolone and a single infusion of rituximab was unsuccessful for both diseases, but the following treatments with 3 courses of rituximab alone for once a week dramatically improved the PRCA as well as the lymphoma. As PRCA complicated with lymphoma is rare, the ideal therapeutic strategy has not yet been established for it. Rituximab may be a good alternative for treating such cases.

[Acquired hemophilia developing after treatment of idiopathic interstitial pneumonia].

A 72-year-old man was diagnosed as having idiopathic interstitial pneumonia (IIP) in May 1999. Immunosuppressive therapy successfully controlled the activity of the IIP. In February 2004, he was referred to our department because of multiple large hematomas. Laboratory examination revealed prolonged activated partial thromboplastin time (APTT; 103.4 seconds), reduced factor VIII activity (7.0%), and the presence of factor VIII inhibitor. Immunosuppressive therapy (prednisolone 1 mg/kg/day) was initiated. After 41 days, APTT decreased to 33.4 seconds and the factor VIII inhibitor disappeared. This is the first reported case of acquired hemophilia which developed during treatment of IIP.

[Successful treatment with drainage of hematoma and chemotherapy in a case of Burkitt leukemia presenting with subdural hematoma].

A 53-year-old male was admitted because of pancytopenia and chronic subdural hematoma. Bone marrow was hypercellular with 97% blasts, which expressed CD10, CD19, CD20, and immunoglobulin mu and gamma chains on the cell surface and had chromosomal abnormalities including t(8 ; 22)(q24 ; q11). The patient was diagnosed as having Burkitt leukemia. Because hemiplegia and disturbance of consciousness developed rapidly, the patient was treated with an emergency drainage operation followed by Hyper-CVAD therapy and achieved a sustained complete remission. Dural infiltration of leukemic cells as well as thrombocytopenia was implicated in the pathogenesis of the subdural hematoma, which contained numerous blasts.