Introduction: Burkitt’s lymphoma (BL) is an endemic malignant neoplasm with a mandibular localisation, first described, in 1958, in African children. In paranasal BL, maxillary sinus is most commonly involved and spheno-ethmoidal sinuses are less commonly involved. The aim of this study, we describe here a rare case of BL in the right maxillary sinus. Case Report: A 23-year-old female presented with a history of right nasal obstruction after pregnancy. In maxillary CT scan a hetrogenous large solid mass showed in the right maxillary sinus which expands this sinus and extends into the right nasal cavity and right ethmoid. She underwent surgical biopsy. In pathology of right maxillary sinus mass biopsy consistent with high grad diffuse small non cleaved NHL. She treated with combination chemotherapy regimen of CODOX-MVAC with triple intrathecal four courses suggested for her. Then she treated with maintenance therapy with thioguanin and MTX and now she is alive for 3 years after last treatment without any new drug and off all treatment and only is in follow up. Conclusion: BL in the right maxillary sinus is very rare but we will have a more successful treatment if it is diagnosed early.
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