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Maryem Ferjani,1 Yosra Zaimi,2 Nouha Trad,2 Yousra Hammi,1 Myriam Ayari,2 Shema Ayadi,2 Taha Sayari,1 Tahar Gargah1 1Department of Pediatrics, Charles Nicolle Hospital, Tunis, Tunisia; 2Department of Gastroenterology, Charles Nicolle Hospital, Tunis, TunisiaCorrespondence: Nouha Trad, Department of Gastroenterology, Charles Nicolle Hospital, Boulevard of April 9, 1938, Bab Saadoun, Tunis, 1007, Tunisia, Tel +216 50858104, Email nouha1med7@gmail.comBackground: Nephrotic syndrome (NS) is associated with a hypercoagulable state and may be complicated by thrombotic events. Venous thrombosis is well-acknowledged, while arterial thrombosis is rather unusual.Case Presentation: We present the case of a 20-year-old woman with a 12-year history of idiopathic NS revealed by extensive cerebral venous thrombosis with pulmonary embolism treated with anticoagulation therapy and oral corticosteroid therapy followed by mycophenolate mofetil (MMF). The thrombophilia assessment did not show any abnormalities. The evolution was marked by the occurrence of several NS relapses controlled by oral corticosteroid therapy until 2017. Subsequently, the patient had not presented a relapse of her disease. The anticoagulant treatment and the MMF were therefore stopped. One year later, the patient presented with severe diffuse acute abdominal pain associated with postprandial vomiting and bilateral lower limb edema. Laboratory results confirmed a NS relapse. An abdominal CT scan revealed acute thrombosis of the superior mesenteric artery with acute mesenteric ischemia. Intraoperative exploration showed mesenteric ischemia with extensive necrosis of the small intestine making their resections incompatible with life. The patient died after 48 hours.Conclusion: Mesenteric arterial thrombosis, which is a rare but life-threatening NS complication, should always be considered, especially in the case of acute non-specific digestive symptoms.Keywords: nephrotic syndrome, mesenteric ischemia, recurrence