Chyle fistula is not a well-known complication of axillary dissection in patients with breast cancer. Although rare, this complication can occur as a result of anatomic variation of the thoracic duct and its venous anastomosis. Injury to the lateral terminating branches or lymphatic trunk, leading to retrograde chyle flow, is more likely than direct injury to the duct. We report a case of chylous fistula after axillary dissection in a patient with breast cancer, review the literature, and discuss the management of this rare complication.