Ischemic colitis (IC) is a rare adverse effect of antipsychotic medications and is most commonly associated with the phenothiazine class of neuroleptics. Different cases reported in patients without other obvious risk factors led to the link between taking neuroleptics and acute ischemic colitis. The severe form is acute necrotizing colitis. This entity is characterized by sudden onset of abdominal pain and bloody diarrhea, progressing rapidly to produce severe illness with general peritonitis and shock.We report a case of a 26 years old Moroccan man, treated for four years for chronic psychosis, admitted to the emergency for abdominal pain and diarrhea. Clinical examination showed a conscious patient, tachycardia at 120 beats/min, febrile to 38.5 ° C, with generalized abdominal defense. Laboratory tests revealed: GB 33400, CRP 290 mg/l, abdominal tomography revealed colonic distension. The patient was prepared and admitted to the operating room. During the intervention, a colonoscopy was performed and shown ulcerated lesions with a purplish background without interval healthy mucosa. A subtotal colectomy with ileostomy and sigmoidostomy were performed. The histological examination of the surgical specimen showed superficial and extended ulcerations without interval healthy mucosa. Thus, no factors for IC were detected by appropriate workup other than the long-time use of neuroleptics. The restoration of continuity by ileorectal anastomosis was achieved two months later with a good clinical outcome, and the patient was recommended for psychiatry to reevaluate his antipsychotic regimen given the association with IC.Our case supports that neuroleptics can promote IC in patients under antipsychotic medications. It should alert physicians who prescribe neuroleptics and colorectal surgeons to the possibility of intestinal ischemia. Although the clinical presentation is non-specific, abdominal pain and distension should be headed, and endoscopy carried out. A better knowledge of this condition should promote earlier diagnosis and improve management.