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Sinonasal lymphomas are relatively uncommon and represent less than 1% of all head and neck malignancies. It is absolutely rare in the United States but its incidence has been reported to be higher in Asian and South American. A primary malignant lymphoma in the paranasal sinuses usually occurs in the maxillary or ethmoid sinus, and it is very rare in the sphenoid sinus. We present a case of a 48-year-old male patient, who complained both of reduced visual acuity and diplopia for a week. An opthalmologic examination revealed no light perception of the right eye. A nasal endoscopy revealed yellowish crusts and a mucosal swelling in both nasal cavity, but no definite mass like lesion was found in the nasal cavity. CT scans of sinuses demonstrated mass lesions in the posterior ethmoid and sphenoid sinus and on the suprasellar area. A mass showed up in the T1 and T2 intensified MR image. He was required to get biopsy for definite diagnosis at the operating room by endoscopic technique, which then allowed it to be pathogenically characterized as angiocentric immunoproliferative lesion (AIL). The lesion was finally defined as T-cell lymphoma by immunochemical study, which showed it to be positive to CD45Ro. After the patient underwent mass biopsy, he received steroid therapy for 5 days. And then both visual acuity was improved abruptly. The patient was treated with both chemotherapy (CHOP) and radiation therapy (4400 cGy). He has been in complete remission state for 1 year after a combination therapy. (Korean J Otolaryngol 2003;46:984-9)