Abstract
Familial hemiplegic migraine (FHM) is a rare, autosomal dominant subtype of migraine characterized by transient hemiparesis and neurological symptoms. It is primarily associated with mutations in the CACNA1A, ATP1A2, and SCN1A genes, which disrupt neuronal excitability and contribute to complex clinical presentations. Although psychotic symptoms accompanying migraine attacks in FHM are exceedingly rare, they have been documented in a limited number of cases. This article presents a case of FHM diagnosed in a patient who exhibited transient psychotic symptoms during a migraine attack, with a focus on diagnostic and therapeutic approaches. The case is discussed in comparison to other rare instances reported in the literature. Psychotic symptoms in FHM are thought to arise from mechanisms such as cortical spreading depression and cerebral hypoperfusion. These symptoms are typically short-lived and respond rapidly to antipsychotic treatment. However, careful evaluation is essential to differentiate these episodes from primary psychotic disorders, thereby avoiding unnecessary prolonged antipsychotic use. This case underscores the importance of a thorough clinical assessment in FHM patients presenting with atypical psychiatric manifestations. The article aims to contribute to the understanding and management of psychotic symptoms in FHM, emphasizing the need for a multidisciplinary approach to diagnosis and treatment. Further research is warranted to elucidate the pathophysiology of psychosis in FHM and to optimize therapeutic strategies for this rare but clinically significant presentation. Keyword: Case report, cortical spreading depression, familial hemiplegic migraine, psychotic disorders.