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Background: Priapism is a urologic emergency representing a true disorder of penile erection that persists beyond or is unrelated to sexual interest or stimulation. Variety of drugs is known to produce priapism, including psychotropics. Case description: We report a case of an adolescent young male with psychosis who developed priapism after taking a single oral dose of 10mg aripiprazole. Discussion: Virtually all antipsychotics have been known to be rarely associated with priapism. The potential of antipsychotics to cause priapism is believed to be dependent on their affinity to block alpha-1 adrenergic receptors. Aripiprazole has the least affinity to adrenergic receptors among all atypical antipsychotics. This makes it an extremely rare occurrence all around the world. Conclusion: Polymorphism of alpha-2A adrenergic receptor gene in schizophrenia patients is known to be associated with sialorrhoea while on clozapine treatment. Probably similar polymorphism of alpha-1 adrenergic receptor gene could contribute to its altered sensitivity and resultant priapism.