Central venous stenosis is a rare cause of unilateral breast edema occurring in hemodialysis patients that needs to be differentiated from other differential diagnoses, including, but not limited to, inflammatory breast carcinoma, mastitis, lymphedema, and congestive heart failure. All reports of similar cases in the available literature have described improvement or resolution of the edema after treatment. Herein, we report and discuss the pathophysiology of breast edema formation in a patient who presented with massive left-sided breast edema 7 years after being diagnosed with central venous stenosis. Medical and minimally invasive therapy had not been successful, so she underwent reduction mammoplasty to relieve the symptoms.