Introduction/Objectives: Clostridium difficile is a spore-forming, toxin-producing, gram-positive anaerobic bacterium that causes antibiotic- associated colitis. Up to 25 percent of patients experience recurrent C. difficile infection (CDI) within 30 days of completing treatment. Extracolonic manifestation of CDI are rare, but recent studies suggest toxin-mediated patogenic process involving cardiac, renal and neurologic impairment . Objectives: Review of clinical manifestations and treatment of patient with dyskinesia as a neurological complication of recurrent CDI. Case report: A 63-year-old female with previous history of arterial hypertension and a condition after a transient ischemic attak, presenting with a moderately severe enterocolitis, dehydration, general weakness and temporary involuntary movements of the left foot, left hand and oral dyskinesias. A clinical examination revealed a left-side hemichoreoathetosis. Three weeks earlier oral vancomycin therapy was administered for the first episode of CDI. Diagnosis was based on laboratory, physical and neurological examinations. The laboratory diagnosis of CDI was confirmed by a positive stool test-enzyme immunoassay for C. difficile toxins A i B (reference interval: negative 0, 37). Results: Hematological-biochemical tests registered a mild increase inflammatory markers and neutrophilia. Thyroid function tests, oncomarkers, electrophoresis of serum proteins and vitamin D3 test were normal. Covid 19 was excluded. A positive stool test-enzyme immunoassay for C. difficile toxin A i B (8, 38) was confirmed a second episode of CDI. Ophthalmological examination, ultrasound of the heart, MSCT and MRI of the brain, electromyography, MR angiography of the brain were normal. Cerebrospinal fluid analysis excluded infection with neurotropic microorganisms and the antibody panel for autoimmune encephalitis was negative (anti-NMDAR, anti-AMPA-R1, anti-AMPA-R2, anti-GABABR, anti-LGI1, anti-CASPR2 antibodies). CDFI of the carotid arteries verified stenosis of the ACI on the right 40%. Electroencephalogram was diffuse irregular. Molecular genetic analysis for Huntington disease was negative (triplet CAG repeat numbers :17/23 in HTT gene). The patient was treated with vancomycin in tapering and pulse doses, and haloperidol 2 mg orally 2 times a day and chlonazepam 0, 5 mg orally 2 times a day for three months. Eradication of CDI and complete recovery of neurological deficit was achieved within four months. Conclusions: We presented a patient with recurrent CDI and dyskinesia as a rare neurological complication most likely induced by C. difficile toxin. Other neurological diseases were excluded by extensive diagnostic work-up. Rational use of antimicrobial therapy and adherence to infection control measures are important in reducing the spread of C.difficile.