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Introduction: Protein-losing enteropathy and spontaneous isolated superior mesenteric artery dissection are both rare clinically. Protein-losing enteropathy due to superior mesenteric artery dissection is extremely rare., Patient Concerns: A 46-year-old male with acute abdominal pain and hematochezia was diagnosed with a complete occlusion of the superior mesenteric artery because of dissection. He suffered from diarrhea and hypoproteinemia after an emergency thromboendarterectomy., Diagnoses: Based on laboratory tests and capsule endoscopy inspection, a diagnosis of protein-losing enteropathy was made., Interventions: Endovascular treatment was provided., Outcomes: After stent placement, he quickly recovered without a recurrence of symptoms., Conclusion: Protein-losing enteropathy is a serious complication of an isolated superior mesenteric artery dissection. Restoring the patency of the superior mesenteric artery is keyed for the treatment of this complication.