Practical Implication:Dystonia following a perinatal injury can have a long latency period in decades, sometimes difficult to treat and may not respond to medical therapy, where deep brain stimulation is another treatment option.Background:Dystonia is defined as a movement disorder characterized by sustained or intermittent muscle contractions causing abnormal, often repetitive, movements, postures, or both, and hemidystonia is dystonic posturing involving one side. (1) Our patient had adolescent-onset, persistent, progressive hemidystonia ( Axis 1 ) secondary to contralateral basal ganglia injury at birth (Axis 2 ) according to recent consensus. (1) He presented as an acute hemidystonic attack with rhabdomyolysis (CPK = 6500 IU/L) and involvement of bulbar and respiratory system with no new structural lesion which is a rare presentation of delayed hemidystonia following perinatal injury.