Introduction: The objective of this case report was to highlight one of the uncommon subtypes of tardive dyskinesia (TD) as tardive blepharospasm secondary to olanzapine. Case Presentation: We reported a rare case of young-onset schizophrenia in an adolescent patient, who was treated with olanzapine, but subsequently developed tardive blepharospasm. Clinical resolution of the blepharospasm after olanzapine was stopped and switched to quetiapine. Conclusions: Tardive blepharospasm can be a regarded as a presentation of tardive dyskinesia. Complete recovery of the symptoms can be achieved by stopping the offending drug early and switching to a serotonin-dopamine receptor antagonist.