Burkitt's lymphoma is an uncommon form of non-Hodgkin's lymphoma (NHL) in adults and represents < 5% of NHL adults. Burkitt's lymphoma involving primarily the appendicular skeleton is rarely described. We present the case of a young man with primary Burkitt's lymphoma involving the humerus as the only site of disease. He received R hyper CVAD and local irradiation and is in complete remission at 24 months.