• Zeming Hu, Xuan Lin, Jianing Zhong, Qingfang He, Qin Peng, Jianbo Xiao, Bin Chen, and Jianhong Zhang.
• Department of General Surgery, The First Affiliated Hospital of Gannan Medical University, Ganzhou, Jiangxi Province, China.
• Medicine (Baltimore). 2020 Jul 10; 99 (28): e21199.

RationaleBlue rubber bleb nevus syndrome (BRBNS) is an extremely rare disorder characterized by multifocal venous malformations involving various organs such as the skin and gastrointestinal tract. Severe complications of BRBNS, such as intussusception, volvulus, and intestinal infarction are rarer and require surgery. This report describes a 33-year-old male of BRBNS complicated with intussusception that was successfully diagnosed and treated with surgery.Patient ConcernsA 33-year-old Chinese man presented with persistent, colicky pain accompanied by nausea, abdominal distension, and dizziness. The patient presented with sporadic bluish nodules on his skin involving his head, neck, thorax, abdomen, and planta pedis.DiagnosesBRBNS with the complication of intussusception.InterventionsAn emergency laparotomy was performed, and postoperative management included blood transfusions and oral iron supplementation for 2 weeks.OutcomesThe patient's postoperative course of hospitalization was uneventful. During the 4-month follow-up, the patient showed no signs of intussusception recurrence.LessonsPatients diagnosed with BRBNS who present with acute abdominal pain and distension should raise suspicion for the presence of intussusception, which requires emergent surgical intervention.

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