: Paroxysmal Hypnogenic Dyskinesia (PHD) is a rare movement disorder characterized by involuntary movements, including chorea, athetosis, ballismus, and dystonia, which occur during the Non-Rapid Eye Movement (NREM) sleep stage. Therefore, the diagnosis of PHD is highly crucial due to the presence of differential diagnoses such as epilepsy and other sleep disorders. Although numerous mutations have been identified, the etiology of PHD, which arises from dysregulation in basal ganglia functions, remains unclear. We wanted to present a case of a nineteen-year-old girl diagnosed with PHD to draw attention to the diagnosis, etiology, and treatment of PHD. The online version contains supplementary material available at 10.1007/s41105-023-00499-5.