References

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Ericson S, Kurol J. Radiographic assessment of maxillary canine eruption in children with clinical signs of eruption disturbance. Eur J Orthod. 1986; 8:133-140 https://doi.org/10.1093/ejo/8.3.133
Richardson G, Russell KA. A review of impacted permanent maxillary cuspids – diagnosis and prevention. J Can Dent Assoc. 2000; 66:497-501
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Peck S, Peck L, Kataja M. Prevalence of tooth agenesis and peg-shaped maxillary lateral incisor associated with palatally displaced canine (PDC) anomaly. Am J Orthod Dentofacial Orthop. 1996; 110:441-443 https://doi.org/10.1016/s0889-5406(96)70048-3
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Unusual Case Report: Ectopic Canines, Resorption and Hypodontia in Monozygotic Twins

From Volume 14, Issue 4, October 2021 | Pages 195-199

Authors

Dayna Rosenthal

BDS (Hons)

Dental Core Trainee in Oral and Maxillofacial Surgery, Royal Free London NHS Foundation Trust

Articles by Dayna Rosenthal

Email Dayna Rosenthal

Paroo Mistry

BDS, MFDS, RCS (Eng), MSc, MOrth RCS (Eng), FDS (Orth) RCS (Eng)

Consultant Orthodontist, Royal Free London NHS Foundation Trust

Articles by Paroo Mistry

Abstract

Ectopic maxillary canines are present in 1–3% of the population and may consequentially cause root resorption in 66.7% of lateral incisors. Ectopic canines are thought to be of polygenic and multifactorial origin, although Msx1 and Pax9 are suggested genetic candidates. Hypodontia is present in up to 11.3% of the population and is associated with mutations in Msx1, Pax9 and Axin2. Previous studies have separately demonstrated similarities in hypodontia and ectopic canines in monozygotic twins. This report discusses similarities clinically and using cone beam computed tomography in 14-year-old male genetically identical twins. While hypodontia and ectopic canines have both previously been linked with a genetic origin, this report suggests there is also a genetic predisposition to root resorption. The association between hypodontia and ectopic canines is usually associated with the guidance theory and hypodontia of upper lateral incisors; however, this report highlights the occurrence of ectopic canines and hypodontia of premolar teeth. It also presents the sequelae of severe root resorption associated with ectopic canines and idiopathically in areas of non-impacted canines

CPD/Clinical Relevance: The role of the general dental practitioner in early recognition of hypodontia and ectopic canines and the importance of referral for an orthodontic opinion is emphasized.

Article

Ectopic maxillary permanent canines occur in 1–3% of the population, over two-thirds of which are impacted palatally.13 Despite the frequency of ectopic canines, they are often missed in routine dental examinations and, as a result, are often not referred until permanent and irreversible resorption has occurred. Although there is no single cause known, two common theories for maxillary canine impaction include the guidance theory and the genetic theory.4 A polygenic, multifactorial genetic pattern of inheritance of palatally displaced canines was suggested by Peck and Peck.5 Similarly, hypodontia is thought to have a multifactorial origin and incidence varies from 2.6% to 11.3% of the population.6 After the wisdom teeth, it most frequently affects the second premolars and lateral incisors. Interestingly, the relationship between ectopic canines and hypodontia has been investigated in several studies, particularly detailing the relationship between ectopic canines and agenesis of lateral incisors based on the guidance theory. There are fewer studies that detail a relationship between palatally ectopic canines and agenesis of the second premolars.79 Furthermore, resorption of incisors adjacent to ectopic canines may be commonly expected sequelae. This report demonstrates the similar presentation of such resorption, but also idiopathic resorption of the lateral incisors on the contralateral side. A genetic link for internal and external root resorption has been suggested in the literature to be the result of genetic polymorphism of the interleukin (IL-1) mutation.10,11 This report of monozygotic twin boys who attended the orthodontic department with palatally ectopic canines, agenesis of one of more second premolars and root resorption of both adjacent and contralateral teeth may, therefore, add to the evidence suggesting a genetic susceptibility for root resorption.

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