The spectrum of psychiatric manifestations in subacute sclerosing panencephalitis: a systematic review of published case reports and case series

Abstract

Background Data related to psychiatric manifestations in SSPE is currently available only in form of isolated case reports. In this systematic review, we evaluated the spectrum of psychiatric manifestations and their impact on the course and outcome of SSPE. Methods Data were obtained from four databases (PubMed, Embase, Scopus, and Google Scholar), with the most recent search conducted on 27/03/2023. The PRISMA guidelines were followed, and the PROSPERO registration number for the protocol is CRD42023408227. SSPE was diagnosed using Dyken's criteria. Extracted data were recorded in an Excel spreadsheet. To evaluate the quality of the data, the Joanna Briggs Institute Critical Appraisal tool was employed. Results Our search resulted in 30 published reports of 32 patients. The mean age was 17.9 years. Schizophrenia, catatonia, and poorly characterized psychotic illnesses were the three most common psychiatric presentations (60%) in SSPE cases. Mania or depression was reported among 23% (7/32) cases. In 10% of cases, the initial clinical presentation of SSPE was considered functional/ hysterical. In approximately 81% (26/32) cases, the course of SSPE was rapidly progressive (either acute fulminant or subacute). Treatment with antipsychotic drugs had poor or no response. Out of 17 patients who received antipsychotic drugs 6 patients noted severe extrapyramidal adverse effects, that further deteriorated the clinical condition of the patients. Conclusion Several patients with SSPE inadvertently end up in psychiatric care due to some psychiatric manifestation. Early psychiatric disorders in SSPE are often subtle and diagnosis of SSPE is easily missed.