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Background: Warfarin induced breast necrosis is a rare complication of oral anticoagulant therapy. Although it can be related to protein C, S, and antithrombin III deficiency; the pathogenesis of necrosis is still unknown. Case presentation: We report a case of a 38-year-old woman with extensive left breast necrosis after receiving warfarin for treatment of deep vein thrombosis. Simple mastectomy was performed and the wound was closed secondarily with an abdominal advancement flap. Rivaroxaban was prescribed after discontinuation of warfarin. Conclusion: Although breast necrosis following warfarin usage is uncommon, it should be considered in women presenting with breast symptoms after initiation of warfarin. Early diagnosis and appropriate management are essential to prevent extensive loss of breast tissue.