Objectives: To describe the clinical characteristics and outcomes of cycloserine (CS)-induced psychosis in adults diagnosed with drug-resistant tuberculosis (DR-TB). Materials and methods: A systematic review of case reports was carried out according to PRISMA guidelines. Subsequently, information was extracted concerning sociodemographic variables, clinical characteristics of psychosis, treatment, and clinical outcomes, as well as the quality of the articles using a standardized tool (Joanna Briggs Institute—JBI—Case Reports Tool). Results: Of 3416 articles, 20 reports from seven countries were included, encompassing 22 patients (68.18% male participants, mean age: 31.45 ± 10.88 years). Delusions (68.2%, primarily persecutory) were the most frequent psychotic symptom. The median duration of the psychotic episode was 13 days (interquartile range: 35). Other frequently appearing symptoms in CS-induced psychosis were aggressiveness (68.2%), insomnia (59.1%), hallucinations (54.5%), incoherent/disorganized speech (45.5%), and irritability (45.5%). After antipsychotic treatment (81.81% of the reported cases were treated with at least one antipsychotic), 95.5% presented improvement, while 4.54% died by suicide. Finally, after the quality assessment of studies using the JBI tool, 85% of the articles showed a low risk of bias. Conclusions: CS-induced psychosis is a rare presentation, generally of short duration, that includes delusions (mostly persecutory) as its main psychotic symptom and shows mostly a symptom improvement after medical treatment. Trial registration PROSPERO registration number: CRD42022359551 (Date of registration: 22/09/2022) © 2023, The Author(s).