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Background. Malignant lymphoma of the testis involves the adjacent epididymis in approximately 60% of cases, but isolated epididymal lymphoma is rare. Methods. The file of a 34-year-old man was reviewed, and the biopsy material was studied by conventional morphology, immunohistochemistry, and polymerase chain reaction. Results. The patient presented with painless bilateral epididymal enlargement. Sequential bilateral epididymal biopsies revealed follicular lymphoproliferation with minimal involvement of one testis. Immunoglobulin M antibodies to the Coxsackie B4 virus were increased, and the lesions were interpreted as an atypical reactive process. Chemotherapy was not pursued. Four years after presentation, there was no evidence of local disease, but a 3-cm lesion involving the skin of the forehead developed. An excision biopsy revealed a large cell follicular lymphoma with features resembling the epididymal lesion. Analysis by polymerase chain reaction revealed a t(14-18) translocation in skin and epididymal lesions confirming that the epididymal lesions were also lymphomatous. Staging revealed no other site of disease. To date, the patient, who was treated with methotrexate chemotherapy, remains well 28 months after treatment. Conclusions. Most lymphomas of the testis and epididymis occur in older men, have an intermediate or high grade, diffuse histology, disseminate early, and follow an aggressive clinical course. In contrast, this case is similar to the occasional reports of pseudolymphoma and follicular lymphoma that occur in younger men, are confined largely to the testis and epididymis, and have a good prognosis. Cancer 1995 ;75 :2174-9.