BACKGROUND Recurrent cystic echinococcosis (CE) with vital organ involvement is a challenge for clinicians. Herein, we report a case of aortic involvement in recurrent retroperitoneal CE lesions following primary splenic CE resection. CASE SUMMARY A 60-year-old male was admitted due to progressive abdominal pain and poor appetite. He was diagnosed with multiple recurrent CE with abdominal aortic involvement according to preoperative evaluation. During surgical resection, major aortic bleeding accidentally occurred while dissecting the cyst, which was firmly attached to the abdominal aortic wall. Hemostasis attempts were conducted to deal with this emergency situation and maintain circulation. Post-interventional recovery was uneventful, and 2-year follow-up showed no sign of recurrence or any other complications. CONCLUSION Radical resection of recurrent complicated CE with aortic involvement should be carefully planned and performed to prevent possible severe adverse complications, thereby improving the postoperative outcome.