Primary cutaneous EBV (Epstein‐Barr virus)‐positive diffuse large B‐cell lymphoma, not otherwise specified (DLBCL, NOS), is an extremely rare disease. The clinical and pathological features of DLBCL, NOS have not been clearly illustrated. We report a case of primary cutaneous EBV‐positive DLBCL, NOS in a 35‐year‐old Chinese male with multiple ulcerated and nodular lesions on his trunk and arms for 6 months. No other organs, except the skin, were involved. The lesions were localized in the dermis with focal necrosis. The tumor cells were immunoblastic‐ or centroblastic‐like cells and diffusely distributed. There were numerous inflammatory cells in the background. The tumor cells were positive for CD20, PAX‐5, MUM1, LMP1, CD30, and Epstein‐Barr virus encodedsmall RNA, but negative for EBNA2. Polymerase chain reaction showed a monoclonal IG gene rearrangement. The patient received 6 cycles of CHOP (cyclophosphamide, hydroxydaunorubicin, Oncovin®, prednisone) chemotherapy and went into complete remission. There was no evidence of relapse after 35 months of follow‐up.
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