One case of allopurinol-caused rare adverse reactions was reported. One male 51-year-old patient presented blurred vision, streaming eyes, photophobia and blepharospasm sequentially 1 week after oral administration of allopurinol. Complete remission was obtained after Botulinum toxin was locally injected. Allopurinol may cause Meige syndrome-like blepharospasm, the mechanism of which may be related to the inhibition of dopamine activity by affecting adenosine level in the brain.