Abstract
BACKGROUND: Foreign language syndrome is a rare neuropsychiatric phenomenon typically following general anesthesia. To date, foreign language syndrome has not been associated with neuroleptic malignant syndrome (NMS) in the literature. This case aims to broaden the clinical understanding of NMS by presenting an atypical manifestation of foreign language syndrome and emphasizing the need for prompt recognition of such presentations for accurate diagnosis and management. CASE PRESENTATION: A 34-year-old Caucasian male with a history of schizoaffective disorder and recurrent psychiatric hospitalizations was admitted for a depressive episode. His condition worsened hours after the administration of intramuscular chlorpromazine, leading to NMS characterized by agitation, muscle rigidity, hyperthermia, autonomic instability, abnormal laboratory findings, and altered mental status, including foreign language syndrome. Management included the discontinuation of the prior psychopharmacotherapy, intravenous hydration, and medications (biperiden, lorazepam). The patient showed significant improvement, with resolution of NMS symptoms and normalized sleep patterns by the time of discharge. CONCLUSION: Foreign language syndrome is an exceptionally rare occurrence, with only nine documented cases to date, all involving male patients. This case presents a novel instance of foreign language syndrome in the context of NMS in a male patient, providing insight into the potential sex-specific mechanisms underlying this rare phenomenon. This case adds valuable evidence to the understanding of the clinical spectrum of NMS and highlights the importance of recognizing atypical presentations in managing patients with neuropsychiatric conditions.