A 23-year-old man developed painful legs and moving toes (PLMT) syndrome while receiving flupentixol/melitracen [Deanxit].The man presented with intermittent movement of his left toes accompanied by prickly pain in his toes and lower leg and a dull ache extending up his thigh. He reported that the pain had preceded abnormal toe movements by 3 months but afterwards, the movements increased in proportion to the pain and became continuous. His abnormal movements were more prominent in supine position and were unchanged with rest or walking; however, they stopped when he was asleep. Additionally, his abnormal movements progressively worsened during the day and were aggravated by stressors, sleep deprivation and caffeine intake. They intensified when his bladder was full and were relieved after emptying the bladder. A detailed medical history revealed that 3 months prior to the development of his symptoms and signs, he had been started on flupentixol/melitracen 0.5/10mg 2 tablets/day [route not stated] for anxiety.The man's flupentixol/melitracen was discontinued; however, his symptoms and signs persisted. On physical examination, abnormal movements were limited to the three middle toes of his left foot and comprised of purposeless writhing with repetitive abduction-adduction. He was able to stop the movements for a short period of time through conscious effort; however, they resumed as soon as he was distracted. He demonstrated an abnormal reaction to cold pressors and light stroke on his left leg. He felt a sharp pricking sensation when his leg was rubbed up to the thigh, or lightly stroked. On cold stimulation, an increase in pain akin to sharp electric shocks with increase in his abnormal movements was noted. He was diagnosed with PLMT syndrome and treated with carbamazepine and botulinum toxin. Subsequently, his abnormal movements significantly reduced and mild improvement in his pain was noted. After treatment discontinuation, the beneficial effect lasted for about 3 months after which his movements and pain gradually reverted to their initial state. At 4 year follow-up, his condition remained stable.Author comment: "[W]e speculate that [antipsychotics] might induce the symptoms of PLMT syndrome".Azzi J, et al. Neuroleptics as a cause of painful legs and moving toes syndrome.