RATIONALE:: Up to one third of all schizophrenic patients are classified as having treatment-resistant schizophrenia (TRS). This subgroup faces remarkable medical and psychosocial damages, and pharmacotherapy is often limited due to nonresponse and/or side effects. Maintenance electroconvulsive therapy (M-ECT) might be effective in TRS. PATIENT CONCERNS:: We present a case of a 26-year-old male patient with a TRS. DIAGNOSES:: He received a treatment series of ECT sessions and a course of 24 M-ECTs. INTERVENTIONS:: The entire treatment was tolerated without significant side effects. OUTCOMES:: Moreover, the Psychotic Symptom Rating Scale (PSYRATS) scores for both positive and negative symptoms decreased and remained stable over the course of M-ECT. LESSONS:: Because of the remarkable improvement in the negative and positive symptom clusters, we propose systematic examinations in the field of M-ECT in TRS patients. These studies should integrate long-term outcome and tolerance measurements, gaining insight into the optimal duration of treatment for this indication.