BACKGROUND A primary central nervous system lymphoma (PCNSL) presenting with massive hemorrhage is a rare occurrence that is difficult to distinguish from a high-grade glioblastoma. Comprehensive descriptions of the imaging characteristics of such tumors have not yet been reported. Herein, we reported a case of a PCNSL with massive hemorrhage by presenting the imaging features of computed tomography (CT) imaging and structural and perfusion magnetic resonance imaging (MRI). METHODS A 48-year-old man presented with headache lasting for 10 d. CT of the brain showed a round, heterogeneous, high-density lesion with surrounding edema in the right temporal lobe. For further diagnosis, a series of MRI examinations of the brain were subsequently performed, and a hemorrhagic lesion with ring-like enhancement was determined. The whole lesion was relatively hypoperfused on arterial spin labeling images. Surgical resection of the lesion and histopathological examination confirmed that the lesion was a diffuse large B-cell lymphoma with massive hemorrhage. CONCLUSIONS PCNSLs with hemorrhage occur very rarely, and structural and perfusion MRI examinations are requested exceedingly rarely. This case provided insight into some characteristics of a hemorrhagic lymphoma on CT and MRI examinations. Perfusion MRI examination may be useful for the differential diagnosis of PCNSLs and other brain tumors.