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Abstract Background Sildenafil citrate is a commonly used medication for the management of erectile dysfunction. Previous studies have described some neuropsychiatric side effects of this medication. So far, however, there has been little discussion about sildenafil-induced psychosis. Case presentation We here present the case of a 32-year-old Iranian male, without a known psychiatric history, who developed psychotic symptoms following initiation of sildenafil. We also postulate a mechanism by which this may occur. Conclusions This report highlights the importance of watchful observation for the occurrence of this rare but serious side effect. Further studies are needed to clarify the precise mechanism that causes sildenafil-induced psychosis.