Cuspid gigantism or radicomegaly is a rare occurrence often associated with a hereditary, X-linked condition, called oculo-cardio-facio-dental (OFCD)syndrome. This syndrome is also characterized by atrial septal defects and/or ventricular septal defects, congenital cataracts and unusual facial characteristics.Confirmation of this syndrome is often provided by dentists or orthodontists when they diagnose an extremely large canine root length from the panoramic radiograph. The following case report shows a multidisciplinary approach to the dental treatment of a patient with OFCD syndrome and a canine-first premolar transposition. The orthodontic approach required a mini-plate to achieve significant mesial movement of a gigantic maxillary canine that erupted in close contact with the first molar.