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Abstract Background Primary central nervous system lymphoma (PCNSL) is an uncommon extranodal non-Hodgkin lymphoma. This tumor is common in the fifth to seventh decade and is located commonly in the periventricular region and in the cerebral hemisphere. Corpus callosum is a rare site. We report a case of diffuse large B cell lymphoma with corpus callosum and contralateral side involvement in immunocompetent patient. Case presentation A 50-year-old male was admitted with complaints of headache with one episode of generalized seizure. On examination, the patient was neurologically intact. CEMRI (contrast-enhanced magnetic resonance imaging) brain showed heterogenous enhancing mass lesion in the left frontal and basifrontal region with involvement of rostrum and body of corpus callosum with patchy areas of diffusion restriction and choline peak on MRS (magnetic resonance spectroscopy). Intraoperatively yellowish brown tumor was seen in the left basifrontal and frontal region with involvement of corpus callosum and extension to opposite side. Microscopic and IHC (immunohistochemistry) examination was suggestive of diffuse large cell lymphoma. Postoperative period was uneventful. The patient received chemoradiotherapy in postoperative period. The patient is asymptomatic 6 months after surgery. Conclusion Primary CNS lymphoma is a rare tumor, and corpus callosum with contralateral side involvement is rare.