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1/227. A man with a swollen leg and abnormal globulins.

    A 72-year-old man presented with a two-week history of exertional dyspnea and nonclaudicatory pain and swelling in the right lower leg. Elevating the leg for short periods had not helped. The symptoms had begun soon after he took a nonsteroidal anti-inflammatory drug for right shoulder pain but persisted after he stopped taking the drug. His history included two-pillow orthopnea, but that had not worsened in the last year. He had not experienced chest discomfort. ( info)

2/227. immunoblastic lymphadenopathy-like T-cell lymphoma complicated by multiple gastrointestinal involvement.

    We report a rare case of immunoblastic lymphadenopathy (IBL)-like T-cell lymphoma complicated by multiple gastrointestinal involvement, which appeared to be ameliorated by chemotherapy but resulted in perforative peritonitis. A 66-year-old Japanese woman who had generalized lymphadenopathy and eruptions was admitted to our hospital because of bloody stool. Colonoscopic examination revealed hemorrhagic ulcers in the terminal ileum and a saucer-like ulcer in the cecum. Gastrointestinal endoscopy revealed several ulcerative or elevated lesions in stomach and duodenum. biopsy specimens of these lesions and of a lymph node showed characteristic histological features of IBL-like T-cell lymphoma. The initial treatment with prednisolone (PSL) and cyclophosphamide (CPA) was effective. Six months after the treatment, however, she developed bloody stool again caused by multiple ulcerative lesions in the large intestine. The recurrence of the disease was determined histologically, and four courses of CPA, PSL, vinblastine sulfate and doxorubicin hydrochloride (CHOP) therapy were administered. One month after completing the CHOP therapy, she developed intestinal obstruction and then acute peritonitis resulting from perforation at an ulcer scar in the jejunum. Surgical treatment was successful, and histological examination demonstrated no lymphoma cells in the resected specimen. A gastrointestinal perforation should be recognized as a potential complication of IBL-like T-cell lymphoma, even during remission. ( info)

3/227. Successful treatment with fludarabine in two cases of angioimmunoblastic lymphadenopathy with dysproteinemia.

    Angioimmunoblastic lymphadenopathy with dysproteinemia (AILD) is today recognized as a T-cell lymphoma which in most cases runs an aggressive course. The diagnosis is often difficult because of the varying clinico-pathological picture. Less than a third of the patients can be expected to have long-term remissions even after multiagent chemotherapy. Complete remissions have been reported after the use of interferon-alpha, cyclosporin A, and recently purine analogues in a few patients. We now report two cases of AILD that had unmaintained remissions for 32 and 10 months, respectively, after fludarabine therapy. In one of the patients fludarabine was used up-front and in the other after she had proved to be resistant to CHOP treatment. No severe infectious complications were noted. The use of purine analogues should be investigated further in AILD. ( info)

4/227. A case of angioimmunoblastic lymphadenopathy with dysproteinemia (AILD) together with total atelectasis of a lung caused by lymphadenopathy and pleural effusion.

    Angioimmunoblastic lymphadenopathy with dysproteinemia (AILD) is a clinicopathologic entity established by Frizzera in 1974. Reported cases of AILD with pleuropulmonary involvement have been increasing recently. At Nogata Central Hospital, an 82-year-old male inpatient with brain infarctions and chronic bronchitis showed a rapid growth of systemic lymphadenopathy and various other symptoms. We diagnosed the case as AILD by histopathologic examination of a lymph node. Chest radiography and computed tomography demonstrated a loss of volume of the right lung caused by intrathoracic lymphadenopathy and a pleural effusion. Although cases of AILD with pleuropulmonary involvement have been increasing, no cases with almost total atelectasis of a lung have been reported as yet. AILD should be taken into account as a disease which may cause atelectasis of a lung. ( info)

5/227. Seropositive rheumatoid arthritis with dermatomyositis sine myositis, angioimmunoblastic lymphadenopathy with dysproteinemia-type T cell lymphoma, and B cell lymphoma of the oropharynx.

    Angioimmunoblastic lymphadenopathy with dysproteinemia (AILD) is a rare lymphoproliferative disorder that often progresses to high grade T cell lymphoma. We describe a 63-year-old woman with longstanding seropositive rheumatoid arthritis who developed fever, cutaneous findings of dermatomyositis, a diffuse pruritic maculopapular rash, enlarged lymph nodes, polyclonal elevated serum gammaglobulins, and an IgG lambda paraprotein. Lymph node biopsies yielded tissue with characteristic changes of AILD and T cell lymphoma. Interleukin 6 (IL-6) was present during the early, active phase of disease, and circulating IL-6 and IL-2 were detected one month before tumor recurrence. Two years after AILD and T cell lymphoma were diagnosed, she developed a B cell lymphoma that involved the oropharynx. ( info)

6/227. Angioimmunoblastic T-cell lymphoma (AIL-TCL) following macrolide administration.

    Angioimmunoblastic Lymphadenopathy with Dysproteinemia (AILD) is a rare benign reactive process which often follows exposure to certain drugs such as penicillin. Treatment with corticosteroids usually reverses the process, however there have been reports of 18% of cases evolving into non-Hodgkins lymphoma. In our case report, we present a relatively healthy woman with history of various drug hypersensitivities who developed AILD and resultant lymphoma after treatment with azithromycin. A review of the literature has failed to find reports of AILD following macrolide exposure. Clonality, not present in other forms of hyperplasia, is present in AILD and immunosuppression may account for this difference. It is difficult to say whether the drugs are simply coincidently associated or actually cause, maintain, or exacerbate clonality in AILD and facilitate malignant transformation. ( info)

7/227. Successful treatment of IBL-like T-cell lymphoma with cyclosporin A: two case reports with special reference to serum cytokine levels.

    The prognosis of immunoblastic lymphadenopathy (IBL)-like T-cell lymphoma is grave, and its effective treatments have not been established. We applied oral cyclosporin A (CsA) treatment to two cases of IBL-like T-cell lymphoma, and succeeded in achieving complete remissions. CsA is known to have a suppressive effect on the immune system, most notably T-cells, but it also has a direct cytotoxic/apoptosis-inducing effect on lymphocytes. Its combined effects on neoplastic T-cells might have played an important role in achieving remission. In both cases, serum levels of interleukin-12 (IL-12) and tumor necrosis factor-alpha (TNF-alpha) were elevated and decreased or returned to normal after achieving remissions. Considering that both cytokines represent monokines, it seems that a macrophage system is also involved in the pathogenesis of this disorder. Our two cases indicate that administration of CsA may be an effective therapy for IBL-like T-cell lymphoma. ( info)

8/227. sulfasalazine-induced angioimmunoblastic lymphadenopathy developing in a patient with juvenile chronic arthritis.

    We describe a patient with juvenile chronic arthritis who developed reactive angioimmunoblastic lymphadenopathy, induced by sulfasalazine. Development of angioimmunoblastic lymphadenopathy although rare, is a very serious side effect of sulfasalazine treatment, and patients treated with this drug should be watched carefully. ( info)

9/227. Immunoblastic sarcoma with features of sjogren's syndrome and systemic lupus erythematosus in a patient with immunoblastic lymphadenopathy.

    A patient with immunoblastic lymphadenopathy and features of sjogren's syndrome and systemic lupus erythematosus is presented. Clinical features included generalized lymphadenopathy, rash, alopecia, and synovitis, with associated laboratory abnormalities of a positive antinuclear factor and double-stranded dna antibodies, positive lupus band test, and hemolytic anemia. Symptoms of sjogren's syndrome included dry eyes and mouth and swollen parotid gland; biopsy results of the accessory salivary glands were positive. At autopsy immunoblastic sarcoma was found that involved the myocardium, which stained for both kappa and lambda light chains by immunoperoxidase techniques. ( info)

10/227. High-dose chemotherapy and APSCT as a potential cure for relapsing hemolysing AILD.

    Angioimmunoblastic lymphadenopathy with dysproteinemia (or dysgammaglobulinemia) (AILD) is a lymphoproliferative disorder with cytogenetic and molecular abnormalities characteristic of malignant T-cell lymphoma (angioimmunoblastic T-cell lymphoma -- AITL). We report the clinical course of a 58-year-old male patient with unusually aggressive AILD, including severe hemolysis and guillain-barre syndrome, who entered complete remission after CHOP therapy, but had a full relapse after 2 months. At relapse, treatment with high-dose chemotherapy followed by autologous peripheral stem cell transplantation (APSCT) with CD34 selected cells was shown to be successful. The patient is alive and disease-free 3 years after diagnosis and 32 months after APSCT. Considering the poor prognosis of the majority of patients with AILD, intensive treatment followed by APSCT, may be a subject for further studies. ( info)
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