Acrocyanosis is probably the most unusual side effect of tricyclic antidepressant drugs. In this paper, we report a 10-year-old boy with primary enuresis nocturna and attention deficit hyperactivity disorder who developed two episodes of acrocyanosis, the first of which was after imipramine treatment and the second after desipramine usage. We also review the possible pathogenesis of acrocyanosis. We suggest that physicians should be aware of this rare side effect of tricyclic antidepressants.