AbstractHypodontia is the congenital absence of less than six teeth because of agenesis. Congenital absence of tooth (hypodontia) from the dental arch, may occur with any tooth, most commonly being third molars, however, absence of permanent mandibular canine is rare. The absence of teeth may be unilateral or bilateral. There are reports showing unilateral occurrence of permanent mandibular canines but agenesis of bilateral mandibular canines is not well documented in the literature and comprehensive review of literature shows paucity of data pertaining to this anomaly. Here we report one such rare case of congenitally missing mandibular permanent canines bilaterally.